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For peer review only

Internet delivery of intensive speech and language therapy for children with cerebral palsy: A pilot randomised

controlled trial

Journal: BMJ Open

Manuscript ID bmjopen-2018-024233

Article Type: Research

Date Submitted by the Author: 17-May-2018

Complete List of Authors: Pennington, Lindsay; Newcastle University, Institute of Health and Society Stamp, Elaine; Newcastle University, Institute of Health and Society Smith, Johanna; Newcastle University, Institute of Health and Society

Kelly, Helen; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language Therapy Parker, Naomi; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language Therapy Stockwell, Katy; Newcastle University, Institute of Health and Society Aluko, Patricia; Newcastle University, Institute of Health and Society Othman, Mohammad; Newcastle University, Open Lab Brittain, Katie; Northumbria University, Department of Nursing, Midwifery & Health Vale, Luke; Newcastle University, UK

Keywords: Speech pathology < OTOLARYNGOLOGY, Developmental neurology & neurodisability < PAEDIATRICS, Clinical trials < THERAPEUTICS

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Internet delivery of intensive speech and language therapy for children with cerebral palsy: A pilot

randomised controlled trial

Lindsay Pennington1, Elaine Stamp

1, Johanna Smith

1, Helen Kelly

2, Naomi Parker

1, Katy Stockwell

1,

Patricia Aluko1, Mohammad Othman

3, Katie Brittain

4, Luke Vale

1

Corresponding author: Lindsay Pennington, Sir James Spence Institute, Royal Victoria Infirmary,

Newcastle upon Tyne, UK, NE1 4LP. Email: [email protected]. Telephone: 0191 282 1360

1 Institute of Health and Society, Newcastle University, Newcastle upon Tyne, UK

2 Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UK

3 Open Lab, Newcastle University, Newcastle upon Tyne, UK

4 Department of Nursing, Midwifery & Health, Northumbria University, Newcastle upon Tyne, UK

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ABSTRACT

Objectives: To test the feasibility of recruitment, retention, outcome measures and delivery of

dysarthria therapy for young people with cerebral palsy in a randomised controlled trial.

Design: Mixed methods. Single blind pilot randomised controlled trial. Qualitative study of the

acceptability of therapy delivery via Skype.

Setting: Nine speech and language therapy departments in northern England recruited participants

to the study. Experimental therapy was provided in a university setting.

Participants: Twenty two children (14 M, 8 F) with dysarthria and cerebral palsy (mean age 8.8 years

(SD 3.2)) agreed to join the study. Participants were randomised to dysarthria therapy via Skype

(n=11) or treatment as usual (n=11).

Interventions: Children received either usual speech therapy from their local therapist for six weeks

or dysarthria therapy via Skype from a research therapist. Usual therapy sessions varied in

frequency, duration and content. Skype dysarthria therapy focussed on breath control and

phonation to produce clear speech at a steady rate, and comprised three 40-minute sessions per

week for six weeks.

Primary and secondary outcome measures: Feasibility and acceptability of the trial design,

intervention and outcome measures.

Results: All participants agreed to random allocation. All completed the study. Recordings of

children’s speech were made at all time points and rated by listeners. Children allocated to Skype

dysarthria therapy and their parents judged the therapy to be acceptable. They reported changes in

the intelligibility of children’s speech, increases in social participation and self-confidence. All

families reported that the study design was acceptable. Treatment integrity checks suggested that

the phrases practiced in one therapy exercise should be reduced in length.

Conclusions: A delayed treatment design, in which dysarthria therapy is offered at the end of the

study to families allocated to treatment as usual, is acceptable. Results suggest that a randomised

controlled trial of dysarthria therapy is feasible.

Article summary

Strengths and limitations of this study

The study addresses a topic which has been identified as a high priority for research by parents and

clinicians.

Assessors of the primary outcome measure were blind to timing of data collection and participant.

The study used a mixed methods approach to assess feasibility and acceptability of the intervention

and study design.

Participants needed to have access to the internet and a computer/tablet at home to be eligible to

join the trial, which may limit the generalisability of the findings.

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INTRODUCTION

Around 20% of children with cerebral palsy have motor disorders affecting speech production,1

causing the speech disorder dysarthria. Dysarthria in cerebral palsy can lead to shallow, uncontrolled

breathing for speech; breathy, harsh voice; reduced pitch variation/unexpected pitch breaks; hyper-

nasality; poor articulation and difficulties in modulating pitch, loudness and timing to vary

intonation.2-5

These characteristics reduce the intelligibility of children’s speech, with severe impacts

on quality of life,6 7

social participation,8 9

education and employment prospects.10

Therapy to improve speech is limited11

but is a priority for families 12

. Small scale studies of therapy

to control respiratory effort for speech and coordinating exhalation with phonation to generate a

clearer speech signal and longer utterances are promising. However, to date, no randomised

controlled trials (RCTs) of this type of intervention for children with cerebral palsy have been

completed.13

The limited evidence base focuses on face to face provision of therapy. Therapy sessions may be

difficult to schedule in school given the number required to promote motor learning (18-20 sessions

over four to six weeks, depending on the therapy programme 14-16

). Attending clinics outside school

hours may not be possible because children with cerebral palsy have considerable mobility

restrictions, and parents have work and other commitments. Web-based therapy is both acceptable

and feasible to other client groups, including people with Parkinson’s and children who stammer 17-

19. If it were found acceptable and feasible to children with cerebral palsy then it would increase

their access to intensive therapy.

This study aimed to examine the acceptability of internet delivery of dysarthria therapy and to test

the feasibility of conducting a RCT comparing intensive dysarthria therapy targeting respiration,

phonation and speech rate delivered via the internet using Skype in increasing the speech

intelligibility of children with cerebral palsy and dysarthria with usual therapy. The objectives were

to ascertain: recruitment rate, adherence to the dysarthria therapy protocols in both randomised

arms, attrition rate, outcome measure completion, data quality, acceptability of the study design to

participant children and their families; and to develop a tool to measure therapy costs. Our previous

research sought to capture natural variation and change in speech by recording children on two

separate occasions twice before and three points after therapy.[14,15] This study additionally aimed

to determine the minimum amount of data that should be collected in a full trial.

DESIGN

Mixed methods study including a single blind, randomised control trial comparing intensive

dysarthria therapy targeting respiration, phonation and speech rate delivered via the internet using

Skype with usual therapy and a qualitative investigation of the views of children and their parents

about the acceptability of the Skype dysarthria therapy and the trial design. The research was given

a favourable opinion by Leeds East Research Ethics Committee.

METHODS

Settings

Eight National Health Service (NHS) speech and language therapy departments in Northern England

and one independent school participated in the study. All provided services to children with cerebral

palsy and were typical of services which would be invited to take part in a full trial.

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Patient involvement

Parents of children with speech difficulties associated with cerebral palsy advised on the overall

design of the study and selected the primary outcome measure of speech intelligibility. A member of

the research team (a parent of a child with cerebral palsy) designed materials for the project and

determined strategies for interaction with parents and children, including recruitment. She also

created the interview topic guides, and led the dissemination of findings to families through a group

workshop and written summaries.

Participants

We aimed to recruit 24 children with cerebral palsy and dysarthria to the study based on the

recommendation of Julious 20

. These indicate that 12 subjects per group is reasonable to consider

feasibility and precision of parameter estimates.

Inclusion criteria: diagnosis of cerebral palsy made by paediatrician or paediatric neurologist;

diagnosis of dysarthria made by speech and language therapist (SLT); parents rate children’s speech

as ‘imprecise but usually understandable to unfamiliar listeners in context’ or ‘unclear and not

usually understandable to unfamiliar listeners out of context’21

; aged 5-17 years; attending schools in

North of England; internet access at home; parents agree to be randomised to either experimental

dysarthria therapy delivered via Skype (herein Skype dysarthria therapy) or usual local therapy with

the offer of Skype dysarthria therapy at the end of the trial.

Exclusion criteria: bilateral hearing loss >50 dB (from prior audiological testing); severe visual

impairments not correctable with glasses (from prior vision testing); unable to understand

grammatically simple instructions (tested by SLTs). Children meeting these criteria have previously

received the therapy.

Intervention

Prior to Skype therapy parents accepted the therapist’s Skype contact request and taught their child

to recognise the therapist’s Skype picture on their PC or tablet. Smart phones were not used as

images of pictures used in therapy would be too small. Parents were with the child when the

therapist called to start each session. Skype dysarthria therapy was delivered three times per week

for six weeks following the Speech Systems Approach.15 16

In this programme sessions last

approximately 40 minutes. Children are taught to control exhalation and phonation to produce a

clear voice in open vowels by copying the therapist’s model. Children are given external targets

relating to how voice sounds, individualised to children’s vocal characteristics e.g. ‘say it in a loud/

big/ strong/ steady voice’, until the most helpful prompt is found. Children then practice using their

(loud, steady, strong) target voice in a hierarchy of speech tasks involving increasingly longer

utterances, from short single words to multisyllabic words and phrases in speaking tasks involving

progressively greater cognitive processing (e.g. conversation, question and answers, guess who

games). Children are required to produce their target voice in 8/10 productions in a task to move to

the next level of the hierarchy. In phrases, children are encouraged to limit the number of syllables

they spoke on a breath and to maintain a speaking rate which sustains the target voice and speech

clarity. In each session children practice using their target voice in at least 120 utterances in four

exercises: (1) 10 open vowels; (2) three productions of 10 self-selected phrases that they often use

in daily life (herein familiar phrases), (3) 70-80 words and phrases from the speech task hierarchy

(e.g. 70 single syllable words); and (4) random presentation of 10-20 utterances from the three

preceding tasks. Feedback is provided frequently to help children acquire the new voice at each level

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and then faded to aid retention. Children are also encouraged to monitor their own productions and

to use internal feedback (e.g. “How did that sound/feel?”).

Usual therapy comprised the therapy children were provided by their local therapy services, at usual

dose and intensity for six weeks.

Recruitment

Children were recruited via SLTs working in participating NHS Trusts and adverts placed in

newsletters from parent support agencies (e.g. Contact, local authority Disabled Children’s

Networks), an independent school for children with disabilities, and parent-carer forums. Children

who appeared to fit the inclusion criteria were visited at home with their parents. Assessments of

speech (Diagnostic Evaluation of Articulation and Phonology22

, Verbal Motor Production Assessment

for Children23

), receptive language (Test of Reception of Grammar 2nd Edition24

) and nonverbal

cognition (Raven’s Coloured Progressive Matrices25

) were undertaken to confirm inclusion.

Adolescents aged 16 years or over and parents provided written consent to participate. Younger

children provided verbal or written assent. All recruitment activities, the time taken and any reasons

given for declining the study were logged.

Outcome measures

Primary outcome

The aim of dysarthria therapy is to increase children’s speech intelligibility. We measured the

intelligibility of children’s speech in single words and connected speech. As in our previous studies,

we elicited single words using the Children’s Speech Intelligibility Measure,26

which comprises 200

lists of 50 single words matched in length and complexity. In this assessment the child repeats words

spoken by the researcher. We developed random allocation schedules to ensure that each list was

allocated a maximum of twice in the study and no list was allocated to the same child twice. We

elicited connected speech using picture description. Children described pictures and the researcher

transcribed their speech verbatim, and then repeated the utterances to check their understanding

with the child. We recorded children’s single word and connected speech at home on two separate

days at six and one week prior to therapy and at one, six and twelve weeks after therapy

completion. Recordings were made using an EDIROL 9 digital recorder, with a table mounted

microphone (Audio Technica Pro24) placed on a table 80 cm from the child (measured at 180°).

To calculate average percentage intelligibility we played each recording to three unfamiliar adults

who had no experience of conversing with people who have speech disorders. We recruited listeners

via adverts in newsletters for personnel in regional businesses. Each listener heard two-three

different recordings via a secure web platform and each recording was heard by three different

listeners. They were instructed not to turn up the volume of the recordings (although this could not

be enforced). In the single word task listeners heard a word and selected the word from a written list

of 12 phonetically similar foils. In the connected speech task listeners orthographically transcribed

each phrase they heard. The system constrained playing of recordings so that each word/phrase

could be played only once. We provided no information about the participants other than that they

had cerebral palsy and were taking part in a study about their speech. We calculated percentage

intelligibility scores for each single word recording and each connected speech recording heard by

each listener using the word lists and gold standard transcriptions created by the researchers when

making the connected speech recordings as the denominators.

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Secondary outcomes

We measured the wider impacts of intervention using the FOCUS27

, a validated measure of

communicative performance and participation. Parents and teachers completed the Parent form

and Clinician form respectively at one week before therapy and twelve weeks after its completion.

Children and parents independently rated the improvements in children’s speech using a seven

point scale: 1 indicates speech intelligibility is almost the same, hardly any better at all; 7 indicates

speech is a very great deal better28

. Text in the children’s scale was accompanied by smiley faces,

graduating in size, and numbers to indicate improvements.

Randomisation

Children were randomly allocated to Skype dysarthria therapy or usual treatment with the offer of

Skype dysarthria therapy at the end of the study in a 1:1 allocation ratio, after the first visit to collect

data (six weeks before therapy). Randomisation was undertaken using an independent web-based

allocation system hosted by Newcastle Clinical Trials Unit.

Adherence to therapy protocol

The Skype dysarthria therapy requires that children produce at least 120 speech behaviours in four

exercises per session. We developed a Treatment Integrity Checklist to code the number of

behaviours elicited in each exercise. We randomly selected two sessions of each child’s Skype

dysarthria therapy to be video-recorded. A researcher who was not involved in delivering therapy to

the recorded child completed the treatment integrity checklist from the video-recording. Five of the

video recordings were also independently rated by a second rater.

Acceptability of Skype delivered dysarthria therapy and trial design

Parents and children allocated to the Skype dysarthria therapy group were interviewed three times

(one week before, three weeks into and six weeks after the intervention) about their experiences of

receiving therapy through Skype. In the third interview they were also asked about their experiences

of randomisation and taking part in the study. Parents and children allocated to usual therapy were

interviewed about the study design at the end of the study. All but two children were interviewed

separately to their parent/carer to ensure each participant’s voice was heard. All interviews were

audio recorded and transcribed orthographically.

Development of costing tool

A questionnaire to measure time required to provide therapy to children allocated to both Skype

therapy and treatment as usual. Therapists recorded: the number of sessions provided to a child; the

time taken to prepare, conduct and follow-up each therapy session and content of sessions for

children allocated to treatment as usual. Travel time duration (in 10 minute intervals); and the NHS

salary band of the person conducting these activities was also collected. We also conducted a review

of tools to measure benefits suitable for use in an economic evaluation, focusing on relevance of the

tool to the client group and the availability of a validated method to convert responses in to health

state utilities which in turn would be used to estimate quality adjusted life years – a common metric

used in economic evaluations.

Analysis

We used descriptive statistics to assess feasibility according to the following criteria:

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• 75% identified children agree to be randomised to dysarthria therapy or usual

treatment;

• 75% children allocated to the usual treatment group are retained for the duration of the

study;

• 85% recorded sessions reach criterion of 120 behaviours produced across four

exercises;

• 75% children recruited to the treatment group and their parents rate therapy via Skype

as acceptable and no insurmountable problems are reported by families;

• 67% in the Skype therapy condition and their parents’ rate therapy as at least

somewhat effective at twelve weeks post therapy.

We analysed transcribed interview data using an inductive thematic analysis29

. Interview transcripts

were coded and stored using Nvivo 10.

To determine if it is possible to reduce the amount of data collected we examined the agreement

between intelligibility measures taken on day one and day two of the paired recordings and between

pre-therapy recordings using mixed effects models, with recording pair nested within data collection

point, nested within child to obtain intra-class correlation coefficients. Then we looked at mean

percentage intelligibility for the number of words taken from the CSIM lists. We looked at taking the

first 15, 20, 30 and 40 words to determine if there was a drop in mean percentage intelligibility when

fewer words were used.

RESULTS

Recruitment

Thirty five children were identified and contacted about the study, of whom 22 were recruited (8

female; mean age 8.8 years (SD 3.2)) between October 2014 and October 2015 (Figure 1). Therapists

from three Trusts who had identified participants in our previous studies identified 1, 3 and 4 eligible

children each, of whom 1, 2, 2 respectively were recruited. Therapists from five Trusts that had not

collaborated in previous studies identified 3-5 (median =4) potential participants each, of whom 2-4

participated (median=3).

All families who fitted the study criteria agreed to be randomised to Skype therapy or treatment as

usual with the offer of Skype therapy at the end of the study. Identification of the first participant in

each of the first wave Trusts took 4-64 days and their recruitment took 19-45 days from

identification.

Insert Figure 1 about here

Characteristics of the study population

Table 1 shows the characteristics of the participants in the two groups. There were more boys in the

treatment as usual group, and overall this group had slightly lower receptive language and nonverbal

understanding than the children allocated to the Skype therapy.

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Table 1 Participant characteristics

Skype dysarthria

therapy group (n=11)

Usual therapy

group (n=11)

Sex 5 M; 6 F 9 M; 2F

Mean Age in years (SD) 8.8 (2.1) 8.8 (4.1)

Type of Cerebral Palsy Spastic 6

Dyskinetic 5

Spastic 6

Dyskinetic 3

Ataxic 2

GMFCS median (IQR) 3 (1, 4) 3 (2, 4)

MACS median (IQR) 2 (1, 3) 2 (2, 3)

CFCS median (IQR) 3 (1, 4) 2 (2, 3)

Viking Speech Scale median (IQR) 3 (2, 3) 3 (2, 3)

Mean language comprehension age (TROG2 age

equivalent score)

7.8 (3.3) 5.6 (2.9)

Mean nonverbal understanding age (Raven’s

Progressive Matrices age equivalent score)

6.6 (2.8) 4.9 (2.0)

Mean length of utterance in words 5.2 (1.9) 5.3 (2.8)

Phonemes articulated (from DEAP, /24) 19.9 (4.5) 18.1 (5.6)

Attrition

None of the participants withdrew from the study.

Outcome measure completion

We completed 206 of 220 planned recordings (94%) of children’s speech. Seven recordings, from the

last four children recruited, could not be completed by the cut-off for recordings which was imposed

by the research time frame a further 7 recordings (from four children) could not take place due to

child illness (4) or holidays (3). We recruited 213 listeners over 12 weeks, who rated a mean of three

recordings each (SD 0.15). Parent and teacher compliance with questionnaire (FOCUS) completion

was low: 29/44 parent questionnaires (66%); 26/44 teacher (59%); 34 pre-therapy (77%); 22 post-

therapy (50%).

Table 2 provides a breakdown of measures collected for participants in the Skype dysarthria and

usual therapy groups.

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Table 2 Outcome measures completed for participants in the Skype dysarthria usual therapy

groups

Measure Skype dysarthria

therapy group

Usual therapy

group

Number of recordings made / total possible (%)

6 week pre therapy

1 week pre therapy

1 week post therapy

6 weeks post therapy


22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

17/18 (94.4)

22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

19/19 (94.4)

Number of listeners hearing recordings / total possible (%)

6 week pre therapy

1 week pre therapy

1 week post therapy



57/66 (86.4)

62/66 (93.9)

59/66 (89.4)

58/66 (87.9)

48/54 (88.9)

58/66 (87.9)

61/66 (92.4)

60/66 (90.9)

60/66 (90.9)

53/57 (93.0)

Number of FOCUS questionnaires completed / total

possible (%)

Parents pre therapy

Parents post therapy

Teachers pre therapy

Teachers post therapy

8/11 (72.7)

6/11 (54.5)

8/11 (72.7)

6/11 (54.5)

10/11 (90.9)

5/11 (45.5)

8/11 (72.7)

4/11 (36.4)

Number of parents rating therapy effectiveness / total

possible (%)

1 week post therapy



11/11 (100)

10/10 (100)

9/9 (100)

NA

NA

NA

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Children received a mean of 15 sessions (SD 2.7, median 16, range 11-18). Children missed sessions

because of illness and competing family commitments. Internet connection issues were encountered

in 32/147 sessions (median 2, range 0-10 per child). Therapy time lost to connection problems

ranged from 2-30 minutes per child over their programme of therapy, (median 15; range 0-38).

Longer connection issues were encountered when new devices were used and one session was

abandoned due to Skype issues that affected users globally.

Video recordings of 31 Skype dysarthria therapy sessions (19%) were checked for treatment fidelity.

Agreement between the two raters on the numbers of behaviours observed in each exercise was

high (chi square 397.4 (df 9) p<0.001). Twenty sessions (65%) met the criterion of 120 behaviours

(mean 121, range 74-140). Failure to elicit 120 speech behaviours arose because the functional

phrases selected by five participants for practice in Exercise 2 were long and could not be spoken on

one breath. This curtailed the time left for rapid elicitation of novel words and phrases and random

practice in Exercises 3 and 4. In two sessions Skype connection difficulties reduced time available to

complete the exercises.

Effectiveness of therapy

All children and parents reported that speech had improved following therapy (Table 3).

Table 3 Children and parents’ ratings of improvements in children’s speech at one, six and twelve

weeks after therapy

Median rating of improvement (range)

1 week 6 weeks 12 weeks

Children’s rating 7 (6 – 7) 7 (4 – 7) 7 (5 – 7)

Parent’s rating 5 (2 – 7) 5 (2 – 7) 6 (4 – 7)

Acceptability of Skype delivery of therapy

All children who received the new therapy and their parents reported that internet delivery via

Skype was acceptable. Some had used Skype before to keep in touch with relatives. Children were

positive about therapy via their tablet/computer, seeing it as ‘cool’ (participant, aged 7). Parents

were happy for the children to be contacted by the research therapists via Skype for pre-arranged

sessions once they had accepted the study Skype name as a contact. Internet connections were

generally good and when connections were lost they were usually retrievable within a session. No

adverse effects on the therapeutic relationship were reported; parents and children reported

enjoying therapy and feeling comfortable with the therapist.

“Basically the same. Just on an iPad instead of face to face” Participant aged 13

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Parents reported that therapy delivery via Skype had an unforeseen benefit of promoting children’s

independence; children used their own computer or tablet, chose where to do therapy and some

older participants logged on independently once parents were sure they recognised the Skype

contact used by the researchers for the study. Parents could remain nearby, see what was

happening in therapy and be on hand to help if was required, but could also attend to other

household tasks and spend time with their other children.

“I think she feels, it gives her a bit more independence. It's hers. …….We'll pop our head in

now and again and make sure she's okay. She doesn't need us at all. She can just do it herself

which is great” 6 year old participant’s father

Parents and children also valued being able to have therapy at home; families did not have to rush to

attend appointments or make additional car journeys, which are difficult for families of children with

disabilities, making clinic appointments stressful for children, parents and siblings. Children liked

doing therapy in familiar surroundings.

“It’s nice that he can do it in his own home, just relaxing….it’s not like ‘ Right come on, we

have to go out’ and drive there, in and out of his wheelchair…..it’s just another trip out which

we would rather avoid if you don’t have to do it” 7 year old participant’s mother

Some parents thought that having some of the therapy sessions via the internet at school may

facilitate greater engagement of education staff in children’s therapy and understanding of it.

“I think that would be quite good because then, at least, you'd be able to get a bit more

feedback from the school as to how they think that it's improved on” Mother of 13 year old

participant

The intensity of the therapy was acceptable for the six week’s duration.

“I think at the moment it's fallen at the right time because I mean our only kind of down side

is a Thursday, which I can try and avoid. Just because of her sister’s activities……. Yes we're

alright at the moment for this next term definitely” 7 year old participant’s mother

“Yes it is manageable…. I couldn't say we could do a fourth one. Three is the limit.” 6 year old

participant’s mother

Acceptability of research design

Random allocation to Skype therapy or treatment as usual was acceptable because of the offer of

the new therapy at the end of the study:

“I think if it was just for a study (without offer of new therapy), I think we would have been

less likely to (participate)” Father of 18 year old participant allocated to treatment as usual

“That was the great thing, it didn’t matter when, you were going to get it regardless, so it

was worth it.” Mother of 10 year old participant allocated to treatment as usual

The visit schedule for the collection of data was time consuming, but deemed to be manageable for

the duration of the study.

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“It is a big commitment, yes. If you've got the time, they do make you aware of what time

you're going to need prior to the therapy. So it's not like you're not aware of it.” Mother of

13 year old participant allocated to Skype therapy

“Just the fact that everything has to be quiet, that’s quite tricky with having a three year old

who’s quite noisy; that was quite hard. And the fact that it had to be all in one week as well.

That was quite tricky. But certainly I wouldn’t say that it was a negative thing, it just was

more difficult to fit in really.” Mother of 6 year old participant allocated to treatment as

usual

All families allocated to treatment as usual accepted the offer of Skype dysarthria therapy at the end

of the study. Having therapy immediately after the end of the study period was preferable for both

parents and children allocated to treatment as usual

“It’s going to work out that we’ve had continuous visits, regular visits, and then the therapy

will start for them. It’s actually maybe worked out quite well. It’s not like it’s been forgotten

and then, “Oh,” all of a sudden here’s the therapy from something you did six months ago.”

Mother of two siblings, one allocated to treatment as usual and one to Skype therapy

All parents and all participants reported that having multiple speech recordings made for the study

was acceptable, but the task did become boring. One child reported that she didn’t like being

recorded but quickly got used to it.

“I’m used to it now. It’s not hard for me, it’s easy now. It used to be hard when I first started.

Now it’s getting easier and easier.” 9 year old girl allocated to Skype therapy group

Neither participants nor parents reported any concerns about the recordings being heard by

strangers in order to estimate children’s intelligibility.

“You’ve got to measure the improvement…. There’d be no point in doing it if you weren’t

going to see a difference….. So it has to be somebody who doesn’t know the children or who

have never heard them before” Mother of child aged 8 allocated to treatment as usual group

Most parents also reported that the FOCUS questionnaire was relevant and easy to complete, and

none reported reasons for not returning the data. One mother reported that she had to check the

meaning of some items and was concerned that she couldn’t recall previous ratings and wanted to

report that she thought her child had improved.

“(remember thinking) I’m misinterpreting this. I can’t just fathom out what they’re asking

me. …. I should have taken a photocopy actually of my answers. Just to remember what I’d

put last time” Mother of child aged 8 allocated to treatment as usual group

Harms

No adverse events were notified during the study.

Economic measures

Therapists of all children allocated to treatment as usual and the research therapists completed all

sections of the resources questionnaire. Results suggested that some sections could be simplified

(therapists’ travelling time to see children could be left open for therapists to complete).

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Six children allocated to the treatment as usual group received at least one session (median 1; range

0-7) of speech and language therapy from NHS services in the six weeks experimental therapy

period. Of the six children who did receive input, two were seen for single review sessions only, two

received regular therapy focussed on expressive language and two received therapy targeting

articulation. Therapy sessions were usually 31-40 minutes in duration. Staff salary bands ranged

from 4 (therapy assistant) to 7 (specialist).

The literature review identified 13 articles reporting the use of tools previously used in this client

group to estimate health state utilities. Apart from generic tools developed for use in children e.g.

HUI2©

, EQ-5D-Y, no other suitable tools were identified. A further tool the CHU-9D, whilst not

previously used, was also considered potentially suitable.

Reducing the data set

There were strong intra-class correlations between percentage intelligibility across pairs of

recordings within the same subject at each of the five time points, for single words this was 0.87

95% CI (0.79, 0.91) and connected speech 0.82 95% CI (0.73, 0.89). Intraclass correlations were also

strong between recordings taken at one and six weeks before therapy for single words (0.83, 95% CI

0.7, 0.90) and connected speech (0.73, 95% CI 0.57, 0.86).

Mean percentage intelligibility for the first 20, 30, and 40 words of the CSIM lists was similar to the

full 50-word lists (Table 4). Mean percentage intelligibility decreased when only the first 15 words

were used.

Table 4 Mean and standard deviation of percentage of words understood in the first 15, 20, 30 and

40 words and full 50-word lists from the Children’s Speech Intelligibility Measure

Words in CSIM lists Percentage of words perceived correctly by listeners

Mean (sd) 95% CI

Full 50-word list

First 30 item

First 25 items

First 20 items

First 15 items

50.27 (18.51)

49.78 (19.41)

49.67 (19.84)

49.55 (20.25)

48.45 (21.14)

48.82 - 51.72

48.26 - 51.30

48.11 - 51.22

47.96 - 51.13

46.80 - 50.11

DISCUSSION

Summary of findings

This study aimed to test the feasibility of conducting a randomised controlled trial of the clinical and

cost effectiveness of intensive dysarthria therapy versus usual treatment for children with cerebral

palsy. Results suggest that a randomised controlled trial is feasible and acceptable but that the study

processes and data collection can be simplified to reduce research costs and burden to participants.

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The study design was acceptable to families. Families joined the research because they were seeking

therapy to improve children’s speech intelligibility, which is highly prized. An important component

of feasibility of recruitment is to offer the experimental therapy to families allocated to treatment as

usual at the end of data collection, even though the treatment may turn out not be effective. There

was no loss of precision in estimates of variation of intelligibility when we reduced the number of

words assessed from 50 to 20. Future trials could limit data collected to single recordings of 20 single

words and picture description at one week pre therapy, and one, six and twelve weeks post therapy.

Return of the FOCUS questionnaire was low (approximately 60%) and could possibly be improved if

usual mechanisms of communication (e.g. home-school bags) were used.

Interviews with parents and participants suggest that provision of therapy via Skype is feasible and

acceptable to families. Internet connection issues affected one in five sessions, but were generally

short and resolvable. However, connection difficulties affected some children more than others,

possibly due to broadband speed. Test sessions to establish Skype connection with families and to

confirm computer settings would be advisable in future research and if implementing Skype in

therapy. None of the parents reported any concerns about internet safety and were happy for

therapists to contact the family via Skype to start therapy sessions. Individualised actions to

maximise internet safety would be required in a trial, as recommended for all children30

, and Trusts

would need to assess information governance issues individually. However, Skype is now being

piloted by NHS Trusts as a medium to conduct consultations and advice exists on its use in clinical

settings.31-33

For most children it was possible to complete all therapy exercises within 40 minutes. Problems that

occurred were due to a selecting phrases that were too long to say in one breath. A similar finding

has been noted for adults with dysarthria caused by Parkinson’s disease. This could be readily

avoided in future trials.

Future trials should test the effectiveness of the therapy when delivered via local therapists. If a

study were clustered at the unit of Trust, to avoid contamination between therapists providing

intervention and participants interacting with each other, mean cluster size can be estimated as

three, based on recruitment from Trusts who had not participated in previous studies. We recruited

a small number of participants who were known to NHS speech and language therapy services via

parent networks and support organisations. Advertisement of trials via parental support networks

should be encouraged to reduce gatekeeping.

Limitations

This study recruited participants from eight trusts within one region. Recruitment may not be

representative of Trusts in other parts of the UK. However, to our knowledge no research is being

conducted on children’s speech elsewhere in the UK and there is sparse research internationally.

Families participating in this study used their own laptops/tablets for the remote therapy and may

represent a ‘tech-savvy’ group. Teletherapy in NHS is only feasible to families with a computer and

internet access; consideration would be needed as to whether this approach might widen socio-

economic health inequalities.

All parents rated speech to have improved at least moderately by twelve weeks. Such lack of

variation precludes the calculation of a minimally important difference in speech intelligibility using

anchor-based methods and suggests change in percentage intelligibility should be used to inform

sample size calculation.34

Only one child judged their speech to have improved moderately (level 4);

all others judged their speech to be a (very) great deal better (6 or 7) at all time points. High ratings

may be due to response bias and social desirability. However, children may have experienced change

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that led them to report their speech being a (very) great deal better. Furthermore, they may also

have developed confidence in or from their ability to be understood, as reported in the interviews.

Future research must measure the impact of therapy on children’s participation and well-being as

well as their intelligibility.12

CONCLUSIONS

The study suggests that a randomised controlled trial of dysarthria therapy is feasible, that therapy

can be provided via Skype to children with cerebral palsy and that families would participate in a trial

if all arms of the study included provision of the new therapy at some point. Future trials could use a

smaller data set to measure intelligibility than previously tested, which would reduce the time and

costs required to conduct a trial and reduce the burden of participating in the study for families.

FUNDING STATEMENT

This paper presents independent research funded by the National Institute for Health Research

(NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-

0712-28077). The views expressed are those of the authors and not necessarily those of the NHS,

the NIHR or the Department of Health and Social Care.

AUTHORS’ CONTRIBUTION

LP, ES, LV, KB designed the study. LP coordinated the study and took overall responsibility for the

research. MO developed the website for conducting listening tasks. HK, NP, JS, KS and PO had

responsibility for data collection. HK, NP, KS delivered the therapy. LP, ES, PO, LV, KB, JS analysed

data. LP drafted the paper. All authors have contributed to and approved the final version of the

paper.

CONFLICTS OF INTEREST

The authors declare no conflicts of interest

DATA SHARING STATEMENT

Data can be obtained by emailing the corresponding author

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2014-006233

13. Pennington L, Parker NK, Kelly H, et al. Speech therapy for children with dysarthria acquired

before three years of age. Cochrane Database of Systematic Reviews 2016(7):Art. No.:

CD006937. DOI: 10.1002/14651858.CD006937.pub3.

14. Fox CM, Boliek CA. Intensive voice treatment (LSVT LOUD) for children with spastic cerebral palsy

and dysarthria. Journal of Speech, Language, and Hearing Research 2012;55(3):930-45.

15. Pennington L, Roelant E, Thompson V, et al. Intensive dysarthria therapy for younger children

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17. Theodoros DG, Hill AJ, Russell TG. Clinical and Quality of Life Outcomes of Speech Treatment for

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2016;25(2):214-32. doi: 10.1044/2015_AJSLP-15-0005

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18. Carey B, O'Brian S, Onslow M, et al. Webcam Delivery of the Camperdown Program for

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19. Howell S, Tripoliti E, Pring T. Delivering the Lee Silverman Voice Treatment (LSVT) by web

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22. Dodd B, Hua Z, Crosbie S, et al. Diagnostic Evaluation of Articulation and Phonology Oxford:

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23. Hayden D, Square P. Verbal motor production assessment for children. San Antonio: Harcourt

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24. Bishop DV. Test of Reception of Grammar 2. Oxford: Pearson Assessment 2003.

25. Raven JC. Raven’s Coloured Progressive Matrices. San Antonio, Tx: PsychCorp 1998.

26. Wilcox K, Morris S. Children's speech intelligibility measure. San Antonio: Harcourt Assessment

1999.

27. Thomas-Stonell N, Oddson B, Robertson B, et al. The FOCUS: Focus on the Outcomes of

Communication Under Six Toronto, Ontario: Holland Bloorview Kids Rehabilitation Hospital;

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28. Guyatt GH, Osoba D, Wu AW, et al. Methods to explain the clinical significance of health status

measures. Mayo Clinic Proceedings 2002;77:371-83.

29. Braun V, Clark V. Using thematic analysis in psychology. Qualitative Research in Psychology

2006;3:77-101.

30. (UKCCIS) UCfCIS. Advice on child internet safety 1.0 Universal guidelines for providers 2012

[Available from:

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dvice_on_child_internet_safety.pdf accessed 13 September 2017.

31. Bostock N. GP pilots Skype consultations with patients. GP Online, 2013.

32. NHS England. 2016 [Available from: https://www.england.nhs.uk/2016/01/newham-diabetes-

pilot-scheme/ accessed 13 September 2017.

33. QIPP Digital Technology. A brief guide to Skype remote consultations: NHS QIPP Digital

Technology; 2012 [Available from: https://www.networks.nhs.uk/nhs-networks/qipp-digital-

technology-and-vision/documents/QIPP%20DT%20Skype%20Factsheet%20v1.pdf accessed

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34. Cook JA, Hislop J, Altman DG, et al. Specifying the target difference in the primary outcome for a

randomised controlled trial: guidance for researchers. Trials 2015;16(1):1-7. doi:

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Figure 1. Participant flow

297x420mm (300 x 300 DPI)

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CONSORT 2010 checklist Page 1

CONSORT 2010 checklist of information to include when reporting a randomised trial*

Section/Topic Item No Checklist item

Reported on page No

Title and abstract

1a Identification as a randomised trial in the title 1 Pilot trial

1b Structured summary of trial design, methods, results, and conclusions (for specific guidance see CONSORT for abstracts) 2

Introduction

Background and

objectives

2a Scientific background and explanation of rationale 3

2b Specific objectives or hypotheses 3

Methods

Trial design 3a Description of trial design (such as parallel, factorial) including allocation ratio 3

3b Important changes to methods after trial commencement (such as eligibility criteria), with reasons NA

Participants 4a Eligibility criteria for participants 4

4b Settings and locations where the data were collected 3,4

Interventions 5 The interventions for each group with sufficient details to allow replication, including how and when they were

actually administered

4,5

Outcomes 6a Completely defined pre-specified primary and secondary outcome measures, including how and when they

were assessed

5,6

6b Any changes to trial outcomes after the trial commenced, with reasons NA

Sample size 7a How sample size was determined 4

7b When applicable, explanation of any interim analyses and stopping guidelines NA

Randomisation:

Sequence

generation

8a Method used to generate the random allocation sequence 6

8b Type of randomisation; details of any restriction (such as blocking and block size) 6

Allocation

concealment

mechanism

9 Mechanism used to implement the random allocation sequence (such as sequentially numbered containers),

describing any steps taken to conceal the sequence until interventions were assigned

6

Implementation 10 Who generated the random allocation sequence, who enrolled participants, and who assigned participants to

interventions

6

Blinding 11a If done, who was blinded after assignment to interventions (for example, participants, care providers, those 5

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CONSORT 2010 checklist Page 2

assessing outcomes) and how

11b If relevant, description of the similarity of interventions 6

Statistical methods 12a Statistical methods used to compare groups for primary and secondary outcomes 7

12b Methods for additional analyses, such as subgroup analyses and adjusted analyses 7

Results

Participant flow (a

diagram is strongly

recommended)

13a For each group, the numbers of participants who were randomly assigned, received intended treatment, and

were analysed for the primary outcome

Figure 1

13b For each group, losses and exclusions after randomisation, together with reasons Figure 1 , 8

Recruitment 14a Dates defining the periods of recruitment and follow-up 7

14b Why the trial ended or was stopped 7

Baseline data 15 A table showing baseline demographic and clinical characteristics for each group 8

Numbers analysed 16 For each group, number of participants (denominator) included in each analysis and whether the analysis was

by original assigned groups

8, 9

Outcomes and

estimation

17a For each primary and secondary outcome, results for each group, and the estimated effect size and its

precision (such as 95% confidence interval)

NA Pilot trial

17b For binary outcomes, presentation of both absolute and relative effect sizes is recommended NA Pilot trial

Ancillary analyses 18 Results of any other analyses performed, including subgroup analyses and adjusted analyses, distinguishing

pre-specified from exploratory

10-13

Harms 19 All important harms or unintended effects in each group (for specific guidance see CONSORT for harms) 12

Discussion

Limitations 20 Trial limitations, addressing sources of potential bias, imprecision, and, if relevant, multiplicity of analyses 14,15

Generalisability 21 Generalisability (external validity, applicability) of the trial findings 14

Interpretation 22 Interpretation consistent with results, balancing benefits and harms, and considering other relevant evidence 14,15

Other information

Registration 23 Registration number and name of trial registry NA

Protocol 24 Where the full trial protocol can be accessed, if available NA

Funding 25 Sources of funding and other support (such as supply of drugs), role of funders 15

*We strongly recommend reading this statement in conjunction with the CONSORT 2010 Explanation and Elaboration for important clarifications on all the items. If relevant, we also

recommend reading CONSORT extensions for cluster randomised trials, non-inferiority and equivalence trials, non-pharmacological treatments, herbal interventions, and pragmatic trials.

Additional extensions are forthcoming: for those and for up to date references relevant to this checklist, see www.consort-statement.org.

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For peer review onlyInternet delivery of intensive speech and language therapy

for children with cerebral palsy: A pilot randomised controlled trial

Journal: BMJ Open

Manuscript ID bmjopen-2018-024233.R1


Date Submitted by the Author: 21-Sep-2018

Complete List of Authors: Pennington, Lindsay; Newcastle University, Institute of Health and SocietyStamp, Elaine; Newcastle University, Institute of Health and SocietySmith, Johanna; Newcastle University, Institute of Health and SocietyKelly, Helen; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language TherapyParker, Naomi; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language TherapyStockwell, Katy; Newcastle University, Institute of Health and SocietyAluko, Patricia; Newcastle University, Institute of Health and SocietyOthman, Mohammad; Newcastle University, Open LabBrittain, Katie; Northumbria University, Department of Nursing, Midwifery & HealthVale, Luke; Newcastle University, UK

<b>Primary Subject Heading</b>: Rehabilitation medicine

Secondary Subject Heading: Paediatrics



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Internet delivery of intensive speech and language therapy for children with cerebral palsy: A pilot

randomised controlled trial

Lindsay Pennington1, Elaine Stamp

1, Johanna Smith

1, Helen Kelly

2, Naomi Parker

1, Katy Stockwell

1,

Patricia Aluko1, Mohammad Othman

3, Katie Brittain

4, Luke Vale

1

Corresponding author: Lindsay Pennington, Sir James Spence Institute, Royal Victoria Infirmary,

Newcastle upon Tyne, UK, NE1 4LP. Email: [email protected]. Telephone: 0191 282 1360





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ABSTRACT

Objectives: To test the feasibility of recruitment, retention, outcome measures and internet delivery

of dysarthria therapy for young people with cerebral palsy in a randomised controlled trial.

Design: Mixed methods. Single blind pilot randomised controlled trial. Qualitative study of the

acceptability of therapy delivery via Skype.

Setting: Nine speech and language therapy departments in northern England recruited participants

to the study. Skype therapy was provided in a university setting.

Participants: Twenty two children (14 M, 8 F) with dysarthria and cerebral palsy (mean age 8.8 years

(SD 3.2)) agreed to take part. Participants were randomised to dysarthria therapy via Skype (n=11) or

treatment as usual (n=11).

Interventions: Children received either usual speech therapy from their local therapist for six weeks

or dysarthria therapy via Skype from a research therapist. Usual therapy sessions varied in

frequency, duration and content. Skype dysarthria therapy focussed on breath control and

phonation to produce clear speech at a steady rate, and comprised three 40-minute sessions per

week for six weeks.

Primary and secondary outcome measures: Feasibility and acceptability of the trial design,

intervention and outcome measures.

Results: Trusts recruited two to three participants. All participants agreed to random allocation.

None withdrew from the study. Recordings of children’s speech were made at all time points and

rated by listeners. Families allocated to Skype dysarthria therapy judged internet delivery of the

therapy to be acceptable. All families reported that the study design was acceptable. Treatment

integrity checks suggested that the phrases practiced in one therapy exercise should be reduced in

length.

Conclusions: A delayed treatment design, in which dysarthria therapy is offered at the end of the

study to families allocated to treatment as usual, is acceptable. A randomised controlled trial of

internet delivered dysarthria therapy is feasible.

Article summary


The study addresses a topic which has been identified as a high priority for research by parents and

clinicians.


The study used a mixed methods approach to assess feasibility and acceptability of the intervention

and study design.

Participants needed to have access to the internet and a computer/tablet at home to be eligible to

join the trial, which may limit the generalisability of the findings.

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INTRODUCTION

Approximately two per thousand infants have cerebral palsy,1, 2

of whom 20%have motor disorders

affecting speech production,3 causing the speech disorder dysarthria. Dysarthria in cerebral palsy

can lead to shallow, uncontrolled breathing for speech; breathy, harsh voice; reduced pitch

variation/unexpected pitch breaks; hyper-nasality; poor articulation and difficulties in modulating

pitch, loudness and timing to vary intonation.4-7

These characteristics reduce the intelligibility of

children’s speech, with severe impacts on quality of life,8, 9

social participation,10, 11

education and

employment prospects.12

Therapy to improve speech is limited, but is a priority for families 13

. Children in the UK receive an

average of 20 hours per year to address both communication and speech issues. Intervention often

targets word production through the Nuffield Dyspraxia Programme14

and non-speech oromotor

function, which have no supporting evidence in childhood dysarthria15

. Small scale studies of

therapy to control respiratory effort for speech and coordinating exhalation with phonation to

generate a clearer speech signal and and a steady speech rate have shown increases in intelligibility,

utterance duration and communicative participation16-19

. However, as yet no randomised controlled

trials (RCTs) of this type of intervention for children with cerebral palsy have been reported.20

To date, therapy focussing on breath support and rate has been provided face to face. But sessions

may be difficult to schedule in school given the number required to promote motor learning (18-20

sessions over four to six weeks, depending on the therapy programme 17, 18, 21

). Attending clinics

outside school hours may not be possible because children with cerebral palsy have considerable

mobility restrictions, in addition to parents’ work and other commitments. A recent systematic

review has shown that web-based therapy shows promise as an acceptable, feasible and effective

method of delivery for children with speech and language disorders22

. The activities involved in

dysarthria therapy (e.g. repetition, picture naming, picture description and conversation) are similar

to those used in previously tested internet therapy23, 24

, and thus it may be possible to deliver

dysarthria programmes as teletherapies. If internet delivery was found acceptable and feasible to

children with cerebral palsy it may increase their access to intensive therapy25

.

This study aimed to examine the acceptability of internet delivery of dysarthria therapy and to test

the feasibility of conducting a RCT comparing intensive dysarthria therapy targeting respiration,

phonation and speech rate delivered via the internet using Skype in increasing the speech

intelligibility of children with cerebral palsy and dysarthria with usual therapy. The objectives were

to ascertain: recruitment rate, attrition rate, outcome measure completion, data quality, and

acceptability of the study design to participant children and their families in both randomised arms

of the study; to measure adherence to the treatment protocol to those allocated to Skype therapy

and gauge families’ perceptions of the acceptability of Skype delivery of therapy; and to develop a

tool to measure therapy costs. Our previous research sought to capture natural variation and change

in speech by recording children on two separate occasions twice before and three points after

therapy17, 18

. This study additionally aimed to determine the minimum amount of data that should be

collected in a full trial.

DESIGN

This feasibility study comprised: 1) a single blind, randomised control pilot trial to compare intensive

dysarthria therapy targeting respiration, phonation and speech rate delivered via the internet using

Skype with usual therapy; and 2) a qualitative investigation of the views of children and their parents

about the acceptability of the Skype dysarthria therapy and the trial design.

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METHODS

Settings

Eight National Health Service (NHS) speech and language therapy departments in Northern England

and one independent school participated in the study. All provided services to children with cerebral

palsy and were typical of services which would be invited to take part in a full trial.

Patient involvement

In our initial study of the intervention, therapy was delivered four times per week for four weeks.

Participants recommended the format be changed to three times per week therapy over six weeks26

.

Parents of children with speech difficulties associated with cerebral palsy took part in workshops

prior to the study to advise on the overall design of the study and select the primary outcome

measure of speech intelligibility. A member of the research team (a parent of a child with cerebral

palsy who had completed postgraduate training in health research) designed materials for the

project and determined strategies for interaction with parents and children, including recruitment.

She also created the interview topic guides, and led the dissemination of findings to families through

a group workshop and written summaries.

Participants

We aimed to recruit 24 children with cerebral palsy and dysarthria to the study based on the

recommendation of Julious 27

. These indicate that 12 subjects per group is reasonable to consider

feasibility and precision of parameter estimates.

Inclusion criteria comprised: diagnosis of cerebral palsy made by paediatrician or paediatric

neurologist; diagnosis of dysarthria made by speech and language therapist (SLT); parents rate

children’s speech as ‘imprecise but usually understandable to unfamiliar listeners in context’ or

‘unclear and not usually understandable to unfamiliar listeners out of context’28

; aged 5-17 years;

attending schools in North of England; internet access at home; parents agree to be randomised to

either dysarthria therapy delivered via Skype (herein Skype dysarthria therapy) or usual local therapy

with the offer of Skype dysarthria therapy at the end of the trial.

Exclusion criteria for the study were bilateral hearing loss >50 dB (from prior audiological testing);

severe visual impairments not correctable with glasses (from prior vision testing); unable to

understand grammatically simple instructions (tested by SLTs).

Intervention

Families used their own PC or tablet and webcams (internal or external) for the therapy. No

additional hardware (e.g. external microphones) was provided. Smart phones were not used as

images of pictures used in therapy would be too small to see clearly. Prior to Skype therapy parents

accepted the therapist’s Skype contact request and taught their child to recognise the therapist’s

Skype picture on their PC or tablet. Parents were with the child when the therapist called to start

each session. Skype dysarthria therapy was delivered three times per week for six weeks following

the Speech Systems Approach.17, 18

Therapy sessions were delivered by one of three therapists

employed for the project (HK, NP, KS), depending on availability. The therapists were generalist

paediatric clinicians who were 6 months – four years post certification and who had no prior

experience of providing motor speech interventions or teletherapy. Prior to the research, they were

trained to conduct the programme according to a written protocol by the lead author through

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demonstration, discussion and self-study. Each therapy session lasted approximately 40 minutes.

Children were taught to control exhalation and phonation to produce a clear voice in open vowels by

copying the therapist’s model. Children were given external targets relating to how voice sounds,

individualised to children’s vocal characteristics e.g. ‘say it in a loud/ big/ strong/ steady voice’, until

the most helpful prompt was found. Children then practiced using their (loud, steady, strong) target

voice in a hierarchy of speech tasks involving increasingly longer utterances, from short single words

to multisyllabic words and phrases in speaking tasks involving progressively greater cognitive

processing (e.g. conversation, question and answers, guess who games). Children were required to

produce their target voice in 8/10 productions in a task to move to the next level of the hierarchy. In

phrases, children were encouraged to limit the number of syllables they spoke on a breath and to

maintain a speaking rate which sustains the target voice and speech clarity. In each session children

practiced using their target voice in at least 120 utterances in four exercises: (1) 10 open vowels; (2)

three productions of 10 self-selected phrases that they often use in daily life (herein familiar

phrases), (3) 70-80 words and phrases from the speech task hierarchy (e.g. 70 single syllable words);

and (4) random presentation of 10-20 utterances from the three preceding tasks. Feedback was

provided frequently to help children acquire the new voice at each level and then faded to aid

retention. Children were also encouraged to monitor their own productions and to use internal

feedback (e.g. “How did that sound/feel?”). Parents were free to sit with their child during the

therapy. During the first two or three sessions all parents sat near their child, watched how

therapists elicited speech behaviours and commented on how their child sounded. Therapists also

asked parents to remind children to use their target voice in daily interaction, but did not train them

to use other therapy techniques. Once they were familiar with the therapy routine, some parents

chose to attend to household tasks out of view of the webcam but within calling distance of the child

if any difficulties arose, others continued to sit nearby.

Usual therapy comprised the therapy children were provided by their local therapy services, at usual

dose and intensity for six weeks.

Recruitment

Children were recruited via SLTs working in participating NHS Trusts and adverts placed in

newsletters from parent support agencies (e.g. Contact, local authority Disabled Children’s

Networks), an independent school for children with disabilities, and parent-carer forums. Children

who appeared to fit the inclusion criteria were visited at home with their parents. Assessments of

speech (Diagnostic Evaluation of Articulation and Phonology29

, Verbal Motor Production Assessment

for Children30

), receptive language (Test of Reception of Grammar 2nd Edition31

) and nonverbal

cognition (Raven’s Coloured Progressive Matrices32

) were undertaken to confirm inclusion.

Adolescents aged 16 years or over and parents provided written consent to participate. Younger

children provided verbal or written assent. All recruitment activities, the time taken and any reasons

given for declining the study were logged.

Outcome measures

Primary outcome

The aim of dysarthria therapy is to increase children’s speech intelligibility. We measured the

intelligibility of children’s speech in single words and connected speech. As in our previous studies,

we elicited single words using the Children’s Speech Intelligibility Measure,33

which comprises 200

lists of 50 single words matched in length and complexity. In this assessment the child repeats words

spoken by the researcher. We developed random allocation schedules to ensure that each list was

allocated a maximum of twice in the study and no list was allocated to the same child twice. We

elicited connected speech using picture description. Children described pictures and the researcher

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transcribed their speech verbatim, and then repeated the utterances to check their understanding

with the child. The research therapists recorded children’s single word and connected speech at

home on two separate days at six and one week prior to therapy and at one, six and twelve weeks

after therapy completion. Recordings were made using an EDIROL 9 digital recorder, with a table

mounted microphone (Audio Technica Pro24) placed on a table 80 cm from the child (measured at

180°).

To calculate average percentage intelligibility we played each recording to three unfamiliar adults

who had no experience of conversing with people who have speech disorders. We recruited listeners

via adverts in newsletters for personnel in regional businesses. Each recording was heard by three

different listeners. Each listener was allocated two-three different recordings via a secure web

platform with the proviso that they heard the same child only once. Listeners were blind to the time

of recording (i.e. pre or post therapy). They were instructed not to turn up the volume of the

recordings (although this could not be enforced). In the single word task listeners heard a word and

selected the word from a written list of 12 phonetically similar foils. In the connected speech task

listeners orthographically transcribed each phrase they heard. The system constrained playing of

recordings so that each word/phrase could be played only once. We provided no information about

the participants other than that they had cerebral palsy and were taking part in a study about their

speech. We calculated percentage intelligibility scores for each single word recording and each

connected speech recording heard by each listener using the word lists and gold standard

transcriptions created by the researchers when making the connected speech recordings as the

denominators.

Secondary outcomes

We measured the wider impacts of intervention using the FOCUS34

, a validated measure of

communicative performance and participation. Parents and children’s class or form teachers

(depending on the child’s school) completed the Parent form and Clinician form respectively at one

week before therapy and twelve weeks after its completion.

Children and parents independently rated the improvements in children’s speech using a seven

point scale: 1 indicates speech intelligibility is almost the same, hardly any better at all; 7 indicates

speech is a very great deal better35

. Text in the children’s scale was accompanied by smiley faces,

graduating in size, and numbers to indicate improvements (see Supplementary Material).

Randomisation

Children were randomly allocated to Skype dysarthria therapy or usual treatment with the offer of

Skype dysarthria therapy at the end of the study in a 1:1 allocation ratio, after the first visit to collect

data (six weeks before therapy). Randomisation was undertaken using an independent web-based

allocation system hosted by Newcastle Clinical Trials Unit.


The Skype dysarthria therapy requires that children produce at least 120 speech behaviours in four

exercises per session. At the start of each exercise the therapist gains eye contact, checks the child’s

sitting posture, describes the exercise, provides a model and instructs the child to produce the

behaviour in their target voice (e.g. ‘Tell me what these are. Use your strong voice.’ We developed a

Treatment Integrity Checklist to code the presence of these therapy actions and the number of

behaviours elicited in each exercise (see Supplementary Material). We randomly selected two

sessions of each child’s Skype dysarthria therapy to be video-recorded. A researcher who was not

involved in delivering therapy to the recorded child completed the treatment integrity checklist from

the video-recording. Five of the video recordings were also independently rated by a second rater.

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Parents and children allocated to the Skype dysarthria therapy group were interviewed three times

(one week before, three weeks into and six weeks after the intervention) about their experiences of

receiving therapy through Skype by a researcher who was not involved in therapy provision (JS). In

the third interview they were also asked about their experiences of randomisation and taking part in

the study. Parents and children allocated to usual therapy were interviewed about the study design

at the end of the study. Interviews were semi-structured and followed a topic guide developed for

the study. Questions about the therapy covered perceived benefits and disadvantages of receiving

therapy via Skype, any memorable experiences of the Skype therapy, impact of therapy on

children’s speech and communication, and parents’ encouragement of children to use their new

voice (the latter two topics are unrelated to feasibility and are reported elsewhere36

). Questions

about the study design asked what it was like to be involved in the research, suggestions for making

it easier for families take part, and views about the measures used. All but two children were

interviewed separately to their parent/carer to ensure each participant’s voice was heard. Initial

interviews took place in children’s homes. Subsequent interviews were taken at home or via

telephone/Skype at the families’ convenience. Two children were interviewed at school with their

teaching assistant present. All interviews were audio recorded and transcribed orthographically.

Interviews with children took approximately 35 minutes (range 15-45 minutes) and 45-50 minutes

with parents.


A questionnaire to measure time required to provide therapy to children allocated to both Skype

therapy and treatment as usual. Therapists recorded: the number of sessions provided to a child; the

time taken to prepare, conduct and follow-up each therapy session and content of sessions for

children allocated to treatment as usual. Travel time duration (in 10 minute intervals); and the NHS

salary band of the person conducting these activities was also collected. We also conducted a

literature review of tools to measure benefits suitable for use in an economic evaluation. PubMed,

Embase, Medline, PsycINFO, NHS EED and Evidence Based Medicine Reviews (Ovid) databases were

searched to identify literatures on tools to measure benefits for children with speech, language and

communication disabilities aged 3-18 years. Full-text articles were included with no year limitation

but language restricted to English. The review focused on the relevance of the tool to the client

group and the availability of a validated method to convert responses in to health state utilities

which in turn would be used to estimate quality adjusted life years – a common metric used in

economic evaluations (further details available from the authors).

Analysis

To determine the feasibility of the study design we assessed the recruitment rate, attrition rate,

outcome measure completion, and data quality in both randomised arms and adherence to the

dysarthria therapy protocol in children allocated to Skype therapy. We used descriptive statistics to

assess feasibility and set the following criteria:

• 75% identified children agree to be randomised to dysarthria therapy or usual

treatment;

• 75% children allocated to the usual treatment group are retained for the duration of the

study;

• 85% recorded sessions reach criterion of 120 behaviours produced across four

exercises;

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• 75% children recruited to the treatment group and their parents rate therapy via Skype

as acceptable and no insurmountable problems are reported by families;

• 67% in the Skype therapy condition and their parents’ rate therapy as at least

somewhat effective at twelve weeks post therapy.

Agreement between raters on the adherence to the therapy protocol was assessed using Kappa for

the presence of therapist actions at the start of each exercise (e.g. describing the exercise, providing

a model) and Pearson correlation coefficient for the number of behaviours elicited in each exercise.

As the aim of the study was to determine the feasibility of the study design, we did not examine

change using our clinical outcome measures37

.

To assess acceptability of delivery of the therapy via Skype and acceptability of the study design we

analysed transcribed interview data using an inductive thematic analysis38

. One researcher (JS)

repeatedly read and compared interview transcripts to develop codes to describe meaning within

interviews, and grouped codes into over-arching themes and sub-themes. Verification of the codes

and themes took place through discussion of code examples and potential themes with two other

researchers (KB and LP). Disagreements were resolved through further review of the dataset and

explanation of thinking behind particular coding or themes. Interview transcripts were coded and

stored using Nvivo 10.

In the development of a tool to assess therapy costs we used descriptive statistics (median, range) to

summarise data relating to therapists’ time spent on activities relating to each participant allocated

to treatment as usual and completion of each section of the data collection form.

To determine if it is possible to reduce the amount of data collected, we examined the agreement

between intelligibility measures taken on day one and day two of the paired recordings and between

pre-therapy recordings using mixed effects regression models, with recording pair nested within

data collection point, nested within child to obtain intra-class correlation coefficients39

. We also used

regression models to examine the effect of word list length on estimates of percentage intelligibility.

Lists comprising the first 15, 20, 25 and 30 words were compared with the full 50 words. In the

examination of the effect of word list length, we fitted child as a random effect and list length as a

fixed effect. We used intraclass correlation coefficients to measure agreement between the reduced

lists and the full 50 word list for the full dataset, and for pre-therapy and post-therapy data

separately.

RESULTS

Feasibility

Recruitment

Thirty five children were identified and contacted about the study, of whom 22 were recruited (8

female, 14 male; mean age 8.8 years (SD 3.2)) between October 2014 and October 2015 (Figure 1).

As dysarthria in cerebral palsy is a chronic condition and children remain on SLT caseloads

throughout childhood, we examined identification of participants by services who had recruited

children to previous studies separately from those with whom we had not worked before. Therapists

from three Trusts who had identified participants in our previous research identified 1, 3 and 4

eligible children each, of whom 1, 2, 2 respectively were recruited. Therapists from five Trusts that

had not collaborated in previous studies identified 3-5 (median =4) potential participants each, of

whom 2-4 participated (median=3).

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All families who fitted the study criteria agreed to be randomised to Skype therapy or treatment as

usual with the offer of Skype therapy at the end of the study, surpassing our feasibility criterion of

75%. Identification of the first participant in each of the first wave Trusts took 4-64 days and their

recruitment took 19-45 days from identification.



Table 1 shows the characteristics of the participants in the two groups. There were more boys in the

treatment as usual group, and overall this group had slightly lower receptive language and nonverbal

understanding than the children allocated to the Skype therapy.


Skype dysarthria

therapy group (n=11)

Usual therapy

group (n=11)

Sex 5 M; 6 F 9 M; 2F



Dyskinetic 5

Spastic 6

Dyskinetic 3

Ataxic 2

GMFCS median (IQR) III (I, IV) I (II, IV)

MACS median (IQR) II (I, III) II (II, III)

CFCS median (IQR) III (I, IV) II (II, III)

Viking Speech Scale median (IQR) III (II, III) III (II, III)

Mean language comprehension age (TROG2 age

equivalent score)

7.8 (3.3) 5.6 (2.9)

Mean nonverbal understanding age (Raven’s

Progressive Matrices age equivalent score)

6.6 (2.8) 4.9 (2.0)



GMFCS: Gross Motor Function Classification Scale40

; MACS: Manual Ability Classification Scale41

;

CFCS Communication Function Classification System42

; TROG2; Test of Reception of Grammar 2nd

edition31

; DEAP: Diagnostic Examination of Articulation and Phonology

Attrition

None of the participants withdrew from the study. All children allocated to Skype dysarthria

completed six weeks of intervention. All children remained in the arm to which they were originally

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allocated, surpassing our feasibility criterion of 75%. However, due to the deadline set by our

funding body, four families were unable to complete measures at twelve weeks post therapy.


We completed 206 of 220 planned recordings (94%) of children’s speech. Seven recordings, from the

last four children recruited, could not be completed by the cut-off for recordings which was imposed

by the research time frame. A further 7 recordings (from four children) could not take place due to

child illness (4) or holidays (3). We recruited 213 listeners over 12 weeks, who rated a mean of three

recordings each (SD 0.15). Parent and teacher compliance with questionnaire (FOCUS) completion

was low: 29/44 parent questionnaires (66%); 26/44 teacher (59%); 34 pre-therapy (77%); 22 post-

therapy (50%).

Table 2 provides a breakdown of measures collected for participants in the Skype dysarthria and

usual therapy groups.

Table 2 Outcome measures completed for participants in the Skype dysarthria usual therapy

groups

Measure Skype dysarthria

therapy group

Usual therapy

group


6 week pre therapy

1 week pre therapy

1 week post therapy



22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

17/18 (94.4)

22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

19/19 (94.4)

Number of listeners hearing recordings / total possible (%)

6 week pre therapy

1 week pre therapy

1 week post therapy



57/66 (86.4)

62/66 (93.9)

59/66 (89.4)

58/66 (87.9)

48/54 (88.9)

58/66 (87.9)

61/66 (92.4)

60/66 (90.9)

60/66 (90.9)

53/57 (93.0)

Number of FOCUS questionnaires completed / total

possible (%)

Parents pre therapy

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8/11 (72.7)

6/11 (54.5)

8/11 (72.7)

6/11 (54.5)

10/11 (90.9)

5/11 (45.5)

8/11 (72.7)

4/11 (36.4)

Number of parents rating therapy effectiveness / total

possible (%)

1 week post therapy



11/11 (100)

10/10 (100)

9/9 (100)

NA

NA

NA


Children received a mean of 15 sessions (SD 2.7, median 16, range 11-18). Children missed sessions

because of illness and competing family commitments. Internet connection issues were encountered

in 32/147 sessions (median 2, range 0-10 per child). Therapy time lost to connection problems

ranged from 2-30 minutes per child over their programme of therapy, (median 15; range 0-38).

Longer connection issues were encountered when new devices were used and one session was

abandoned due to Skype issues that affected users globally.

Video recordings of 31 Skype dysarthria therapy sessions (19%) were checked for treatment fidelity.

Agreement between the two raters on the presence of therapy activities at the start of each exercise

was substantial43

(Κ=0.72) and high for the numbers of behaviours produced in each exercise (r

=0.99, p<0.001). Twenty sessions (65%) met the criterion of 120 behaviours (mean 121, range 74-

140), thus failing to meet the feasibility criterion of 85% of sessions containing 120 speech

behaviours. Failure to elicit 120 speech behaviours arose because the functional phrases selected by

five participants for practice in Exercise 2 were long and could not be spoken on one breath. This

curtailed the time left for rapid elicitation of novel words and phrases and random practice in

Exercises 3 and 4. In two sessions Skype connection difficulties reduced time available to complete

the exercises.


All eight children and eight parents who rated effectiveness of therapy at twelve weeks post

intervention deemed it at least moderately effective (median rating by children 5, range 5-7; median

rating by parents 6, range 4-7). . Should the three children and their parents who were unable to

complete ratings due to time limitations have judged it less than somewhat effective, the criterion of

67% rating the therapy as at least somewhat effective would have been reached.

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Acceptability

Skype delivery of therapy

Discussion about the delivery of the intervention via Skype centred around four key themes: 1)

Familiarity with technology; 2) Remote connections; 3) Fit with family life and 4) Unforeseen

benefits.

Familiarity with technology

During the interviews none of the parents raised concerns about using Skype for therapy. Those

allocated to the Skype therapy described themselves as being familiar with using technology for

communication; some had used Skype before to keep in touch with relatives and others had used

alternative video communication software such as Facetime. Parents were happy for the children to

be contacted by the research therapists via Skype for pre-arranged sessions once they had accepted

the study Skype name as a contact.

Skype I haven’t used in probably about five or six years. But yes, we’ve used FaceTime a lot.

(Father A)

I'll show you how to get Skype on and then you'll get that ready. When I tell him, once he's

done it, he'll know how to set it up because he's good with computers, so it won't be a

problem. (Mother E)

Children were also positive about therapy via their tablet/computer, seeing it as ‘cool’ (Child G).

Most appeared confident about using the technology from the start, although one child who had not

used Skype or FaceTime had lots of questions about how to work the software and appeared a little

anxious about being able to connect. However, in post therapy interviews this child reported that

she quickly ‘got used’ to Skype after the first couple of sessions.

I know Skype. Will it ring? (Child Q)

There is a button you have to press, you know the ‘enter’ button on the computer? When

you’ve got the little arrow, and you can move the arrow around with the mouse?

(Interviewer)

I don’t know, yes I do. And you just click the button. What if we don’t have a mouse? I think

it’s going to be better if someone like peeked through the door. If I need help with a hard

question and I peek through the door and say I am struggling, and they come and help me.

(Child Q)

Remote connections

Parents reported that internet connections were generally good and that when connections were

lost they were usually retrievable within a session.

There was one day I think [research therapist] was struggling to connect. She just Skyped a

typed message saying, “I’m just having a few problems.” … But on the whole our Broadband

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has been fine because it does stick a little bit sometimes where we are. But we don’t seem to

have had any issues really. (Mother E)

We never even had any issues with Wi-Fi connection and our Wi-Fi's rubbish. We never got a

lost signal through once, I don't think that's happened once. If there is any, it's just like me

being the idiot and not turning up the volume up. (Father A)

Parents and children reported enjoying therapy and feeling comfortable with the therapist.

Basically the same. Just on an iPad instead of face to face. (Child R)

I think it's great in the fact that it is over Skype. You've not got people coming in all the time,

in and out of the house, which is not a problem. But it makes it a bit more fun for the

youngsters, I think, by having Skype. [Child] was sitting there with his iPad and he was really

enjoying it. (Mother R)

However, one child reported that sitting in the same position for the duration of the Skype sessions

was difficult

It’s difficult to do sitting. On Skype it’s different. (Child V)

On Skype it’s difficult to sit still for that long. (Mother V)

Overall, although shorter sessions might be better for some children, it seemed that the families did

not view the technology as a barrier to therapy.

Fit with family life

Parents and children reported that a key benefit of Skype therapy delivery was being able to have

therapy at home; families did not have to rush to attend appointments or make additional car

journeys, which are difficult for families of children with disabilities, making clinic appointments

stressful for children, parents and siblings. Children liked doing therapy in familiar surroundings.

It’s nice that he can do it in his own home, just relaxing….it’s not like ‘ Right come on, we

have to go out’ and drive there, in and out of his wheelchair…..it’s just another trip out which

we would rather avoid if you don’t have to do it (Mother I)

However, the three times per week schedule was difficult to manage across the full six weeks for

some families.

Yes it is manageable…. I couldn't say we could do a fourth one. Three is the limit. (Mother V)

Some parents thought that having some of the therapy sessions via the internet at school may

facilitate greater engagement of education staff in children’s therapy and understanding of it.

I think that would be quite good because then, at least, you'd be able to get a bit more

feedback from the school as to how they think that it's improved on. (Mother R)

Unforeseen benefits

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Parents reported that therapy delivery via Skype had an unforeseen benefit of promoting children’s

independence. They described how children used their own computer or tablet, chose where to do

therapy and some older participants logged on independently once parents were sure they

recognised the Skype contact used by the researchers for the study. Parents described how they

could remain nearby, see what was happening in therapy and be on hand to help if was required,

but could also attend to other household tasks and spend time with their other children. This meant

that parents could be involved but also let their children take control and ownership of their

therapy.

I think she feels, it gives her a bit more independence. It's hers. …….We'll pop our head in now

and again and make sure she's okay. She doesn't need us at all. She can just do it herself

which is great. (Father A)

Yes, kind of, you’re listening to how well she’s doing, but equally taking in what the girls are

doing with her, so that you can kind of apply some of it at home. (Mother G)

Research design

Discussion around the study design centred on the random allocation to Skype therapy or usual

therapy and the outcome measures used in study. One of the motivating factors in taking part, even

if allocated to usual care, was the offer of therapy via skype at the end, and this seemed crucial for

successful engagement with participants. :

I think if it was just for a study [without offer of Skype therapy], I think we would have been

less likely to (participate). (Father D)

That was the great thing, it didn’t matter when, you were going to get it regardless, so it was

worth it. (Mother O)

The visit schedule for the collection of data was time consuming, but deemed to be manageable for

the duration of the study.

It is a big commitment, yes. If you've got the time, they do make you aware of what time

you're going to need prior to the therapy. So it's not like you're not aware of it. (Mother R)

Just the fact that everything has to be quiet, that’s quite tricky with having a three year old

who’s quite noisy; that was quite hard. And the fact that it had to be all in one week as well.

That was quite tricky. But certainly I wouldn’t say that it was a negative thing, it just was

more difficult to fit in really. (Mother V)

All families allocated to treatment as usual accepted the offer of Skype dysarthria therapy at the end

of the study. Having therapy immediately after the end of the study period was seen as preferable

for both parents and children allocated to treatment as usual

It’s going to work out that we’ve had continuous visits, regular visits, and then the therapy

will start for them. It’s actually maybe worked out quite well. It’s not like it’s been forgotten

and then, “Oh,” all of a sudden here’s the therapy from something you did six months ago.

(Mother F and J)

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All parents and all participants reported that having multiple speech recordings made for the study

was acceptable, but that the task did become boring. One child reported that she didn’t like being

recorded but quickly got used to it.

I’m used to it now. It’s not hard for me, it’s easy now. It used to be hard when I first started.

Now it’s getting easier and easier. (Child C)

Neither participants nor parents reported any concerns about the recordings being heard by

strangers in order to estimate children’s intelligibility.

You’ve got to measure the improvement…. There’d be no point in doing it if you weren’t

going to see a difference….. So it has to be somebody who doesn’t know the children or who

have never heard them before” (Mother E)

Most parents also reported that the FOCUS questionnaire was relevant and easy to complete, and

none reported reasons for not returning the data. One mother reported that she had to check the

meaning of some items and was concerned that she couldn’t recall previous ratings and wanted to

report that she thought her child had improved.

(remember thinking) I’m misinterpreting this. I can’t just fathom out what they’re asking me.

…. I should have taken a photocopy actually of my answers. Just to remember what I’d put

last time (Mother E)

Harms


Economic measures of therapy costs

Therapists of all children allocated to treatment as usual and the research therapists completed all

sections of the resources questionnaire. Results suggested that some sections could be simplified

(therapists’ travelling time to see children could be left open for therapists to complete).

Six children allocated to the treatment as usual group received at least one session (median 1; range

0-7) of speech and language therapy from NHS services in the six weeks therapy period assessed for

the study. Of the six children who did receive input, two were seen for single review sessions only,

two received regular therapy focussed on expressive language and two received therapy targeting

articulation. Therapy sessions were usually 31-40 minutes in duration. Staff salary bands ranged

from 4 (therapy assistant) to 7 (specialist).

The literature review identified 13 articles reporting the use of tools previously used in this client

group to estimate health state utilities. Apart from generic tools developed for use in children e.g.

HUI2©

, EQ-5D-Y, no other suitable tools were identified. A further tool the CHU-9D, whilst not

previously used, was also considered potentially suitable.


There were strong intra-class correlations between percentage intelligibility across pairs of

recordings within the same subject at each of the five time points, for single words this was 0.87

(95% CI 0.79, 0.91) and connected speech 0.82 (95% CI 0.73, 0.89). Intraclass correlations were also

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strong between recordings taken at one and six weeks before therapy for single words (0.83, 95% CI

0.7, 0.90) and connected speech (0.73, 95% CI 0.57, 0.86).

Mixed effect regression models showed very little impact of list length on intelligibility after

adjusting for the fixed effect of child when the full 50 item lists were compared with 30, 25 or 20

items. However, when the first 15 items were compared with the full 50 item list a significant

difference was observed (95% CI -3.87, 0.36). There were strong intra-class correlations between the

20, 25, 30 and 50 words of the CSIM lists , for both the full dataset and when pre-therapy and post

therapy data were examined separately (Table 3).

Table 3 Mean and standard deviation of percentage of words understood in the first 15, 20, 30 and

40 words and full 50-word lists from the Children’s Speech Intelligibility Measure

Words

in CSIM

lists

Percentage of words

perceived correctly by

listeners

Comparison with full 50 word list

All data Pre-therapy Post-therapy

Mean

(sd)

95% CI ICC 95% CI ICC 95% CI ICC 95% CI

Full 50

word

list

50.27

(18.51)

48.82-51.72

- - - - - -

First 30

item

49.78

(19.41)

48.26-51.30

0.97 0.96-0.97 0.97 0.95-0.98 0.97 0.96-0.98

First 25

items

49.67

(19.84)

48.11-51.22

0.96 0.95-0.96 0.96 0.94-0.97 0.96 0.95-0.97

First 20

items

49.55

(20.25)

47.96-51.13

0.93 0.93-0.94 0.92 0.90-0.94 0.94 0.92-0.95

First 15

items

48.45

(21.14)

46.80-50.11 0.89 0.87-91 0.87 0.83-0.90 0.90 0.87-0.92

DISCUSSION

Summary of findings

This study aimed to test the feasibility of conducting a randomised controlled trial of the clinical and

cost effectiveness of intensive dysarthria therapy versus usual treatment for children with cerebral

palsy. Results suggest that a randomised controlled trial is feasible and acceptable but that the study

processes and data collection can be simplified to reduce research costs and burden to participants.

The study design was acceptable to families. Families joined the research because they were seeking

therapy to improve children’s speech intelligibility and communicative independence, which is highly

prized44

. An important component of feasibility of recruitment was the offer of the experimental

therapy to families allocated to treatment as usual at the end of data collection, even though the

treatment may turn out not be effective. Our findings here concur with the results of previous trials

of interventions for children with other types of neurodisability, such as autism45-47

, and support the

conclusion that parents of children with neurodisability often feel the need to ‘do more’ for their

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children continue to seek additional input that could aid their child’s development48, 49

. There was no

loss of precision in estimates of variation of intelligibility when we reduced the number of words

assessed from 50 to 20, which is similar to other tests of intelligibility50

. Future trials could limit data

collected to single recordings of 20 single words and picture description at one week pre therapy,

and one, six and twelve weeks post therapy. Return of the FOCUS questionnaire was low

(approximately 60%) and could possibly be improved if usual mechanisms of communication (e.g.

home-school diaries and bags) were used51

.

Similar to other studies of teletherapy for children with speech disorders and adults with

dysarthria22, 24, 52

, interviews with parents and participants suggest that provision of therapy via

Skype is feasible and acceptable to families. Therapy was delivered according to the therapy

protocol and children and parents reported no difficulties in maintaining rapport with the therapist,

supporting other recent research22, 24, 53

One possible advantage of the study design was that the

research therapists were familiar to families by the time therapy was due to commence, having

carried out the assessments and speech recordings. Shaw and colleagues, in their study of general

practice consultations, found that sessions via video-conferencing flowed more easily when patients

and practitioners knew each other and had time to build a relationship53

. Internet connection issues

affected one in five sessions in our study, but were generally short and resolvable as previous UK

research53

. However, connection difficulties affected some children more than others, possibly due

to broadband speed. Test sessions to establish Skype connection with families and to confirm

computer settings would be advisable in future research and if implementing Skype in therapy53

.

None of the parents reported any concerns about internet safety and were happy for therapists to

contact the family via Skype to start therapy sessions. Individualised actions to maximise internet

safety would be required in a trial, as recommended for all children54

, and Trusts would need to

assess information governance issues individually53

. However, Skype is now being piloted by NHS

Trusts as a medium to conduct consultations and advice exists on its use in clinical settings.55-57

For most children it was possible to complete all therapy exercises within 40 minutes. Problems that

occurred were due to a selecting phrases that were too long to say in one breath. A similar finding

has been noted for adults with dysarthria caused by Parkinson’s disease58

. This could be readily

avoided in future trials. Shortening the phrases may also help to reduce the overall session length,

making them more accessible to children who find it difficult to maintain their concentration.

Therapy was conducted by three generalist clinicians who had no experience of childhood motor

speech disorders or teletherapy. As such, they were similar to many NHS therapists who provide

services to children in mainstream schools in the UK, but less experienced than therapists who

provide specialist services to children with motor disorders15

. This study has shown that the protocol

can be followed by junior therapists and by extension it should be suitable for therapists with

greater experience. Future trials should test the effectiveness of the therapy when delivered via

local therapists, and include a process evaluation to evaluate barriers to and facilitators of

implementation which have not been identified in our research to date59

. If a study were clustered

at the unit of Trust, to avoid contamination between therapists providing intervention and

participants interacting with each other, mean cluster size can be estimated as three, based on

recruitment from Trusts who had not participated in previous studies. We recruited a small number

of participants who were known to NHS speech and language therapy services via parent networks

and support organisations. Advertisement of trials via parental support networks should be

encouraged to reduce gatekeeping.

Limitations

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This study recruited participants from eight trusts within one region. Recruitment may not be

representative of Trusts in other parts of the UK. However, to our knowledge no research is being

conducted on children’s speech elsewhere in the UK and there is sparse research internationally20

.

Families participating in this study used their own laptops/tablets for the remote therapy and may

represent a ‘tech-savvy’ group. Teletherapy in NHS is only feasible to families with a computer and

internet access; consideration would be needed as to whether this approach might widen socio-

economic health inequalities.

Intelligibility measures were conducted by the research therapists, which could have prompted

children to use their new voice during measurement. Future research should ensure that

measurements are conducted by personnel who have not been involved in therapy but who have

built up a rapport with children so that they feel comfortable during in the assessments.

All parents rated speech to have improved at least moderately by twelve weeks. Such lack of

variation precludes the calculation of a minimally important difference in speech intelligibility using

anchor-based methods and suggests change in percentage intelligibility should be used to inform

sample size calculation.60

Only one child judged their speech to have improved moderately (level 4);

all others judged their speech to be a (very) great deal better (6 or 7) at all time points. High ratings

may be due to response bias and social desirability. However, children may have experienced change

that led them to report their speech being a (very) great deal better. Furthermore, they may also

have developed confidence in or from their ability to be understood, as reported in the interviews.

Future research must measure the impact of therapy on children’s participation and well-being as

well as their intelligibility.13

CONCLUSIONS

The study suggests that a randomised controlled trial of dysarthria therapy is feasible, that therapy

can be provided via Skype to children with cerebral palsy and that families would participate in a trial

if all arms of the study included provision of Skype dysarthria therapy at some point. Future trials

could use a smaller data set to measure intelligibility than previously tested, which would reduce the

time and costs required to conduct a trial and reduce the burden of participating in the study for

families.

FUNDING STATEMENT

This paper presents independent research funded by the National Institute for Health Research

(NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-

0712-28077). The views expressed are those of the authors and not necessarily those of the NHS,

the NIHR or the Department of Health and Social Care.

ETHICS STATEMENT

The study was approved by National Research Ethics Service Committee Leeds East Research Ethics

Committee (14/YH/0058).


LP, ES, LV, KB designed the study. LP coordinated the study and took overall responsibility for the

research. MO developed the website for conducting listening tasks. HK, NP, JS, KS and PO had

responsibility for data collection. HK, NP, KS delivered the therapy. LP, ES, PO, LV, KB, JS analysed

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data. LP drafted the paper. All authors have contributed to and approved the final version of the

paper.





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Randomized Controlled Trial. American Journal of Speech-Language Pathology 2016; 25: 214-232.

DOI: 10.1044/2015_AJSLP-15-0005.

59. Craig P, Dieppe P, Macintyre S, et al. Developing and evaluating complex interventions: new

guidance. London: Medical Research Council, 2008.

60. Cook JA, Hislop J, Altman DG, et al. Specifying the target difference in the primary outcome

for a randomised controlled trial: guidance for researchers. Trials 2015; 16: 1-7. journal article. DOI:

10.1186/s13063-014-0526-8.

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297x420mm (300 x 300 DPI)

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Internet delivery of intensive speech and language therapy for children with cerebral palsy

Rating of effectiveness of therapy Parents ‘Is your child’s speech better, about the same or worse than before they started the Skype speech therapy?’ (if better) ‘How much better is your child’s speech? ’

1 2 3 4 5 6 7 Almost the same A little better Somewhat better Moderately better A good deal better A great deal better A very great deal

better If worse ‘How much worse is your child’s speech?’

1 2 3 4 5 6 7 Almost the same A little worse Somewhat worse Moderately worse A good deal worse A great deal worse A very great deal worse Children ‘Is your speech better, about the same or worse than before they started the Skype speech therapy?’ (if better) ‘How much better is your speech? ’

1 2 3 4 5 6 7

If worse ‘How much worse is your speech?’

1 2 3 4 5 6 7

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1

Lindsay Pennington, Newcastle University 2015

FIDELITY TREATMENT INTEGRITY CHECK©

INTENSIVE SPEECH AND LANGUAGE THERAPY FOR CHILDREN WITH CEREBRAL PALSY:

A SYSTEMS APPROACH THERAPY PROTOCOL (ROBSON, MILLER, PENNINGTON 207)

Participant SLT

Date of session

Observer Date of observation

1) TARGET VOICE IN SUSTAINED VOWEL

Production of /a:/

1. Therapist checks child’s position at start of exercise YES/NO

2. Therapist gains child’s visual attention at start of exercise YES/NO

3. Therapist describes exercise: practice ‘ah’ 10 times YES/NO

4. Therapist provides modelling appropriate to phase YES/NO

5. Therapist instructs child e.g. “Copy me/you do it” YES/NO

6. Therapist provides stimulus phrase e.g. “use your [target voice name] voice YES/NO

7. Number of repetitions elicited (/10)

2) OPTIMUM SPEECH LOUDNESS AND RATE

Production of functional phrases



10. Therapist describes exercise YES/NO


12. Therapist instructs child e.g. “You do it/Your turn” YES/NO

13. Therapist provides stimulus phrase e.g. “use your [target] voice” YES/NO

14. 3 repetitions of each functional phrase elicited YES/NO

15. Feedback and modelling for individual attempts

Feedback type: Specific directive Reflective

Feedback frequency: Each behaviour After 1-5 behaviours After 6-10

Feedback: Knowledge of Performance Knowledge of Results

16. Positive reinforcement given YES/NO

3) STIMULATE OPTIMAL LOUDNESS IN NOVEL STIMULI

The child is asked to repeat 80 - 120 stimuli provided by the therapist. Circle stage

Stage a) Single words or phrases Stage b) Phrases Stage c) Extended speech Stage d) Increased cognitive difficulty - describe Stage e) Conversation


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2




20. Therapist provides elicitor appropriate to phase YES/NO

21. Therapist provides stimulus phrase e.g. “use your ‘nice and easy’ voice” YES/NO

22. Number of behaviours elicited (/80)






4) RANDOMISATION OF STIMULI



27. Therapist describes exercise: practice of all targets YES/NO


29. Number of items elicited (/20)

30. Feedback





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CONSORT 2010 checklist of information to include when reporting a pilot or feasibility trial*


Reported on page No

Title and abstract

1a Identification as a pilot or feasibility randomised trial in the title 1

1b Structured summary of pilot trial design, methods, results, and conclusions (for specific guidance see CONSORT abstract extension for pilot trials)

2

Introduction

Background and

objectives

2a Scientific background and explanation of rationale for future definitive trial, and reasons for randomised pilot trial

3

2b Specific objectives or research questions for pilot trial 3

Methods

Trial design 3a Description of pilot trial design (such as parallel, factorial) including allocation ratio 4

3b Important changes to methods after pilot trial commencement (such as eligibility criteria), with reasons NA


4b Settings and locations where the data were collected 4 - 7

4c How participants were identified and consented 5



5

Outcomes 6a Completely defined prespecified assessments or measurements to address each pilot trial objective specified in 2b, including how and when they were assessed

8

6b Any changes to pilot trial assessments or measurements after the pilot trial commenced, with reasons NA

6c If applicable, prespecified criteria used to judge whether, or how, to proceed with future definitive trial 8

Sample size 7a Rationale for numbers in the pilot trial 4


Randomisation:

Sequence

generation


8b Type of randomisation(s); details of any restriction (such as blocking and block size) 6

Allocation

concealment

mechanism

9 Mechanism used to implement the random allocation sequence (such as sequentially numbered containers), describing any steps taken to conceal the sequence until interventions were assigned

6

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interventions

6

Blinding 11a If done, who was blinded after assignment to interventions (for example, participants, care providers, those


6

11b If relevant, description of the similarity of interventions NA

Statistical methods 12 Methods used to address each pilot trial objective whether qualitative or quantitative 8

Results

Participant flow (a

diagram is strongly

recommended)

13a For each group, the numbers of participants who were approached and/or assessed for eligibility, randomly assigned, received intended treatment, and were assessed for each objective

9 and Figure

13b For each group, losses and exclusions after randomisation, together with reasons 10 and Figure


14b Why the pilot trial ended or was stopped NA

Baseline data 15 A table showing baseline demographic and clinical characteristics for each group Table 1

Numbers analysed 16 For each objective, number of participants (denominator) included in each analysis. If relevant, these numbers

should be by randomised group 11, 12

Outcomes and

estimation

17 For each objective, results including expressions of uncertainty (such as 95% confidence interval) for any estimates. If relevant, these results should be by randomised group

12, 12, 16

Ancillary analyses 18 Results of any other analyses performed that could be used to inform the future definitive trial 12-15


19a If relevant, other important unintended consequences 14

Discussion

Limitations 20 Pilot trial limitations, addressing sources of potential bias and remaining uncertainty about feasibility 18

Generalisability 21 Generalisability (applicability) of pilot trial methods and findings to future definitive trial and other studies 18

Interpretation 22 Interpretation consistent with pilot trial objectives and findings, balancing potential benefits and harms, and

considering other relevant evidence 17, 19

22a Implications for progression from pilot to future definitive trial, including any proposed amendments 17

Other information

Registration 23 Registration number for pilot trial and name of trial registry NA

Protocol 24 Where the pilot trial protocol can be accessed, if available Not available


26 Ethical approval or approval by research review committee, confirmed with reference number 19

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Citation: Eldridge SM, Chan CL, Campbell MJ, Bond CM, Hopewell S, Thabane L, et al. CONSORT 2010 statement: extension to randomised pilot and feasibility trials. BMJ. 2016;355.

*We strongly recommend reading this statement in conjunction with the CONSORT 2010, extension to randomised pilot and feasibility trials, Explanation and Elaboration for important

clarifications on all the items. If relevant, we also recommend reading CONSORT extensions for cluster randomised trials, non-inferiority and equivalence trials, non-pharmacological

treatments, herbal interventions, and pragmatic trials. Additional extensions are forthcoming: for those and for up to date references relevant to this checklist, see www.consort-statement.org.

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For peer review onlyInternet delivery of intensive speech and language therapy

for children with cerebral palsy: A pilot randomised controlled trial

Journal: BMJ Open

Manuscript ID bmjopen-2018-024233.R2


Date Submitted by the Author: 16-Nov-2018

Complete List of Authors: Pennington, Lindsay; Newcastle University, Institute of Health and SocietyStamp, Elaine; Newcastle University, Institute of Health and SocietySmith, Johanna; Newcastle University, Institute of Health and SocietyKelly, Helen; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language TherapyParker, Naomi; Newcastle Upon Tyne Hospitals NHS Foundation Trust, Speech and Language TherapyStockwell, Katy; Newcastle University, Institute of Health and SocietyAluko, Patricia; Newcastle University, Institute of Health and SocietyOthman, Mohammad; Newcastle University, Open LabBrittain, Katie; Northumbria University, Department of Nursing, Midwifery & HealthVale, Luke; Newcastle University, UK

<b>Primary Subject Heading</b>: Rehabilitation medicine

Secondary Subject Heading: Paediatrics



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Internet delivery of intensive speech and language therapy for children with cerebral palsy: A pilot randomised controlled trial

Lindsay Pennington1, Elaine Stamp1, Johanna Smith1, Helen Kelly2, Naomi Parker1, Katy Stockwell1, Patricia Aluko1, Mohammad Othman3, Katie Brittain4, Luke Vale1

Corresponding author: Lindsay Pennington, Sir James Spence Institute, Royal Victoria Infirmary, Newcastle upon Tyne, UK, NE1 4LP. Email: [email protected]. Telephone: 0191 282 1360





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mailto:[email protected]



ABSTRACT

Objectives: To test the feasibility of recruitment, retention, outcome measures and internet delivery of dysarthria therapy for young people with cerebral palsy in a randomised controlled trial.

Design: Mixed methods. Single blind pilot randomised controlled trial, with control offered Skype therapy at end of study. Qualitative study of the acceptability of therapy delivery via Skype.

Setting: Nine speech and language therapy departments in northern England recruited participants to the study. Skype therapy was provided in a university setting.

Participants: Twenty two children (14 M, 8 F) with dysarthria and cerebral palsy (mean age 8.8 years (SD 3.2)) agreed to take part. Participants were randomised to dysarthria therapy via Skype (n=11) or treatment as usual (n=11).

Interventions: Children received either usual speech therapy from their local therapist for six weeks or dysarthria therapy via Skype from a research therapist. Usual therapy sessions varied in frequency, duration and content. Skype dysarthria therapy focussed on breath control and phonation to produce clear speech at a steady rate, and comprised three 40-minute sessions per week for six weeks.

Primary and secondary outcome measures: Feasibility and acceptability of the trial design, intervention and outcome measures.

Results: Departments recruited two to three participants. All participants agreed to random allocation. None withdrew from the study. Recordings of children’s speech were made at all time points and rated by listeners. Families allocated to Skype dysarthria therapy judged internet delivery of the therapy to be acceptable. All families reported that the study design was acceptable. Treatment integrity checks suggested that the phrases practiced in one therapy exercise should be reduced in length.

Conclusions: A delayed treatment design, in which dysarthria therapy is offered at the end of the study to families allocated to treatment as usual, is acceptable. A randomised controlled trial of internet delivered dysarthria therapy is feasible.

Article summary


The study addresses a topic which has been identified as a high priority for research by parents and clinicians.


The study used a mixed methods approach to assess feasibility and acceptability of the intervention and study design.

Participants needed to have access to the internet and a computer/tablet at home to be eligible to join the trial, which may limit the generalisability of the findings.

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INTRODUCTION

Approximately two per thousand infants have cerebral palsy,1, 2 of whom 20%have motor disorders affecting speech production,3 causing the speech disorder dysarthria. Dysarthria in cerebral palsy can lead to shallow, uncontrolled breathing for speech; breathy, harsh voice; reduced pitch variation/unexpected pitch breaks; hyper-nasality; poor articulation and difficulties in modulating pitch, loudness and timing to vary intonation.4-7 These characteristics reduce the intelligibility of children’s speech, with severe impacts on quality of life,8, 9 social participation,10, 11 education and employment prospects.12

Therapy to improve speech is limited, but is a priority for families 13. Children in the UK receive an average of 20 hours per year to address both communication and speech issues. Intervention often targets word production through the Nuffield Dyspraxia Programme14 and non-speech oromotor function, which have no supporting evidence in childhood dysarthria15. Small scale studies of therapy to control respiratory effort for speech and coordinating exhalation with phonation to generate a clearer speech signal and and a steady speech rate have shown increases in intelligibility, utterance duration and communicative participation16-19. However, as yet no randomised controlled trials (RCTs) of this type of intervention for children with cerebral palsy have been reported.20

To date, therapy focussing on breath support and rate has been provided face to face. But sessions may be difficult to schedule in school given the number required to promote motor learning (18-20 sessions over four to six weeks, depending on the therapy programme 17, 18, 21). Attending clinics outside school hours may not be possible because children with cerebral palsy have considerable mobility restrictions, in addition to parents’ work and other commitments. A recent systematic review has shown that web-based therapy shows promise as an acceptable, feasible and effective method of delivery for children with speech and language disorders22. The activities involved in dysarthria therapy (e.g. repetition, picture naming, picture description and conversation) are similar to those used in previously tested internet therapy23, 24 , and thus it may be possible to deliver dysarthria programmes as teletherapies. If internet delivery was found acceptable and feasible to children with cerebral palsy it may increase their access to intensive therapy25.

This study aimed to examine the acceptability of internet delivery of dysarthria therapy and to test the feasibility of conducting a RCT comparing intensive dysarthria therapy targeting respiration, phonation and speech rate delivered via the internet using Skype in increasing the speech intelligibility of children with cerebral palsy and dysarthria with usual therapy. The objectives were to ascertain: recruitment rate, attrition rate, outcome measure completion, data quality, and acceptability of the study design to participant children and their families in both randomised arms of the study; to measure adherence to the treatment protocol to those allocated to Skype therapy and gauge families’ perceptions of the acceptability of Skype delivery of therapy; and to develop a tool to measure therapy costs. Our previous research sought to capture natural variation and change in speech by recording children on two separate occasions twice before and three points after therapy17, 18. This study additionally aimed to determine the minimum amount of data that should be collected in a full trial.

DESIGN

This feasibility study comprised: 1) a single blind, randomised control pilot trial to compare intensive dysarthria therapy targeting respiration, phonation and speech rate delivered via the internet using Skype with usual therapy; and 2) a qualitative investigation of the views of children and their parents about the acceptability of the Skype dysarthria therapy and the trial design.

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METHODS

Settings

Eight National Health Service (NHS) speech and language therapy departments in Northern England and one independent school participated in the study. All provided services to children with cerebral palsy and were typical of services which would be invited to take part in a full trial.

Patient involvement

In our initial study of the intervention, therapy was delivered four times per week for four weeks. Participants recommended the format be changed to three times per week therapy over six weeks26. Parents of children with speech difficulties associated with cerebral palsy took part in workshops prior to the study to advise on the overall design of the study and select the primary outcome measure of speech intelligibility. A member of the research team (a parent of a child with cerebral palsy who had completed postgraduate training in health research) designed materials for the project and determined strategies for interaction with parents and children, including recruitment. She also created the interview topic guides, and led the dissemination of findings to families through a group workshop and written summaries.

Participants

We aimed to recruit 24 children with cerebral palsy and dysarthria to the study based on the recommendation of Julious 27. These indicate that 12 subjects per group is reasonable to consider feasibility and precision of parameter estimates.

Inclusion criteria comprised: diagnosis of cerebral palsy made by paediatrician or paediatric neurologist; diagnosis of dysarthria made by speech and language therapist (SLT); parents rate children’s speech as ‘imprecise but usually understandable to unfamiliar listeners in context’ or ‘unclear and not usually understandable to unfamiliar listeners out of context’28; aged 5-17 years; attending schools in North of England; internet access at home; parents agree to be randomised to either dysarthria therapy delivered via Skype (herein Skype dysarthria therapy) or usual local therapy with the offer of Skype dysarthria therapy at the end of the trial.

Exclusion criteria for the study were bilateral hearing loss >50 dB (from prior audiological testing); severe visual impairments not correctable with glasses (from prior vision testing); unable to understand grammatically simple instructions (tested by SLTs).

Intervention

Families used their own PC or tablet and webcams (internal or external) for the therapy. No additional hardware (e.g. external microphones) was provided. Smart phones were not used as images of pictures used in therapy would be too small to see clearly. Prior to Skype therapy parents accepted the therapist’s Skype contact request and taught their child to recognise the therapist’s Skype picture on their PC or tablet. Parents were with the child when the therapist called to start each session. Skype dysarthria therapy was delivered three times per week for six weeks following the Speech Systems Approach.17, 18 Therapy sessions were delivered by one of three therapists employed for the project (HK, NP, KS), depending on availability. The therapists were generalist paediatric clinicians who were 6 months – four years post certification and who had no prior experience of providing motor speech interventions or teletherapy. Prior to the research, they were trained to conduct the programme according to a written protocol by the lead author through

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demonstration, discussion and self-study. Each therapy session lasted approximately 40 minutes. Children were taught to control exhalation and phonation to produce a clear voice in open vowels by copying the therapist’s model. Children were given external targets relating to how voice sounds, individualised to children’s vocal characteristics e.g. ‘say it in a loud/ big/ strong/ steady voice’, until the most helpful prompt was found. Children then practiced using their (loud, steady, strong) target voice in a hierarchy of speech tasks involving increasingly longer utterances, from short single words to multisyllabic words and phrases in speaking tasks involving progressively greater cognitive processing (e.g. conversation, question and answers, guess who games). Children were required to produce their target voice in 8/10 productions in a task to move to the next level of the hierarchy. In phrases, children were encouraged to limit the number of syllables they spoke on a breath and to maintain a speaking rate which sustains the target voice and speech clarity. In each session children practiced using their target voice in at least 120 utterances in four exercises: (1) 10 open vowels; (2) three productions of 10 self-selected phrases that they often use in daily life (herein familiar phrases), (3) 70-80 words and phrases from the speech task hierarchy (e.g. 70 single syllable words); and (4) random presentation of 10-20 utterances from the three preceding tasks. Feedback was provided frequently to help children acquire the new voice at each level and then faded to aid retention. Children were also encouraged to monitor their own productions and to use internal feedback (e.g. “How did that sound/feel?”). Parents were free to sit with their child during the therapy. During the first two or three sessions all parents sat near their child, watched how therapists elicited speech behaviours and commented on how their child sounded. Therapists also asked parents to remind children to use their target voice in daily interaction, but did not train them to use other therapy techniques. Once they were familiar with the therapy routine, some parents chose to attend to household tasks out of view of the webcam but within calling distance of the child if any difficulties arose, others continued to sit nearby.

Usual therapy comprised the therapy children were provided by their local therapy services, at usual dose and intensity for six weeks.

Recruitment

Children were recruited via SLTs working in participating NHS Trusts and adverts placed in newsletters from parent support agencies (e.g. Contact, local authority Disabled Children’s Networks), an independent school for children with disabilities, and parent-carer forums. Children who appeared to fit the inclusion criteria were visited at home with their parents. Assessments of speech (Diagnostic Evaluation of Articulation and Phonology29, Verbal Motor Production Assessment for Children30), receptive language (Test of Reception of Grammar 2nd Edition31) and nonverbal cognition (Raven’s Coloured Progressive Matrices32) were undertaken to confirm inclusion. Adolescents aged 16 years or over and parents provided written consent to participate. Younger children provided verbal or written assent. All recruitment activities, the time taken and any reasons given for declining the study were logged.

Outcome measures

Primary outcomeThe aim of dysarthria therapy is to increase children’s speech intelligibility. We measured the intelligibility of children’s speech in single words and connected speech. As in our previous studies, we elicited single words using the Children’s Speech Intelligibility Measure,33 which comprises 200 lists of 50 single words matched in length and complexity. In this assessment the child repeats words spoken by the researcher. We developed random allocation schedules to ensure that each list was allocated a maximum of twice in the study and no list was allocated to the same child twice. We elicited connected speech using picture description. Children described pictures and the researcher

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transcribed their speech verbatim, and then repeated the utterances to check their understanding with the child. The research therapists recorded children’s single word and connected speech at home on two separate days at six and one week prior to therapy and at one, six and twelve weeks after therapy completion. Recordings were made using an EDIROL 9 digital recorder, with a table mounted microphone (Audio Technica Pro24) placed on a table 80 cm from the child (measured at 180°).

To calculate average percentage intelligibility we played each recording to three unfamiliar adults who had no experience of conversing with people who have speech disorders. We recruited listeners via adverts in newsletters for personnel in regional businesses. Each recording was heard by three different listeners. Each listener was allocated two-three different recordings via a secure web platform with the proviso that they heard the same child only once. Listeners were blind to the time of recording (i.e. pre or post therapy). They were instructed not to turn up the volume of the recordings (although this could not be enforced). In the single word task listeners heard a word and selected the word from a written list of 12 phonetically similar foils. In the connected speech task listeners orthographically transcribed each phrase they heard. The system constrained playing of recordings so that each word/phrase could be played only once. We provided no information about the participants other than that they had cerebral palsy and were taking part in a study about their speech. We calculated percentage intelligibility scores for each single word recording and each connected speech recording heard by each listener using the word lists and gold standard transcriptions created by the researchers when making the connected speech recordings as the denominators.

Secondary outcomesWe measured the wider impacts of intervention using the FOCUS34, a validated measure of communicative performance and participation. Parents and children’s class or form teachers (depending on the child’s school) completed the Parent form and Clinician form respectively at one week before therapy and twelve weeks after its completion. Children and parents independently rated the improvements in children’s speech using a seven point scale: 1 indicates speech intelligibility is almost the same, hardly any better at all; 7 indicates speech is a very great deal better35. Text in the children’s scale was accompanied by smiley faces, graduating in size, and numbers to indicate improvements (see Supplementary Material).

Randomisation

Children were randomly allocated to Skype dysarthria therapy or usual treatment with the offer of Skype dysarthria therapy at the end of the study in a 1:1 allocation ratio, after the first visit to collect data (six weeks before therapy). Randomisation was undertaken using an independent web-based allocation system hosted by Newcastle Clinical Trials Unit.


The Skype dysarthria therapy requires that children produce at least 120 speech behaviours in four exercises per session. At the start of each exercise the therapist gains eye contact, checks the child’s sitting posture, describes the exercise, provides a model and instructs the child to produce the behaviour in their target voice (e.g. ‘Tell me what these are. Use your strong voice.’ We developed a Treatment Integrity Checklist to code the presence of these therapy actions and the number of behaviours elicited in each exercise (see Supplementary Material). We randomly selected two sessions of each child’s Skype dysarthria therapy to be video-recorded. A researcher who was not involved in delivering therapy to the recorded child completed the treatment integrity checklist from the video-recording. Five of the video recordings were also independently rated by a second rater.

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Parents and children allocated to the Skype dysarthria therapy group were interviewed three times (one week before, three weeks into and six weeks after the intervention) about their experiences of receiving therapy through Skype by a researcher who was not involved in therapy provision (JS). In the third interview they were also asked about their experiences of randomisation and taking part in the study. Parents and children allocated to usual therapy were interviewed about the study design at the end of the study. Interviews were semi-structured and followed a topic guide developed for the study. Questions about the therapy covered perceived benefits and disadvantages of receiving therapy via Skype, any memorable experiences of the Skype therapy, impact of therapy on children’s speech and communication, and parents’ encouragement of children to use their new voice (the latter two topics are unrelated to feasibility and are reported elsewhere36). Questions about the study design asked what it was like to be involved in the research, suggestions for making it easier for families take part, and views about the measures used. All but two children were interviewed separately to their parent/carer to ensure each participant’s voice was heard. Initial interviews took place in children’s homes. Subsequent interviews were taken at home or via telephone/Skype at the families’ convenience. Two children were interviewed at school with their teaching assistant present. All interviews were audio recorded and transcribed orthographically. Interviews with children took approximately 35 minutes (range 15-45 minutes) and 45-50 minutes with parents.


A questionnaire to measure time required to provide therapy to children allocated to both Skype therapy and treatment as usual. Therapists recorded: the number of sessions provided to a child; the time taken to prepare, conduct and follow-up each therapy session and the aim and content of sessions for children allocated to treatment as usual. Travel time duration (in 10 minute intervals); and the NHS salary band of the person conducting these activities was also collected. We also conducted a literature review of tools to measure benefits suitable for use in an economic evaluation. PubMed, Embase, Medline, PsycINFO, NHS EED and Evidence Based Medicine Reviews (Ovid) databases were searched to identify literatures on tools to measure benefits for children with speech, language and communication disabilities aged 3-18 years. Full-text articles were included with no year limitation but language restricted to English. The review focused on the relevance of the tool to the client group and the availability of a validated method to convert responses in to health state utilities which in turn would be used to estimate quality adjusted life years – a common metric used in economic evaluations (further details available from the authors).

Analysis

To determine the feasibility of the study design we assessed the recruitment rate, attrition rate, outcome measure completion, and data quality in both randomised arms and adherence to the dysarthria therapy protocol in children allocated to Skype therapy. We used descriptive statistics to assess feasibility and set the following criteria:

75% identified children agree to be randomised to dysarthria therapy or usual treatment;

75% children allocated to the usual treatment group are retained for the duration of the study;

85% recorded sessions reach criterion of 120 behaviours produced across four exercises;

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75% children recruited to the treatment group and their parents rate therapy via Skype as acceptable and no insurmountable problems are reported by families;

67% in the Skype therapy condition and their parents’ rate therapy as at least somewhat effective at twelve weeks post therapy.

Agreement between raters on the adherence to the therapy protocol was assessed using Kappa for the presence of therapist actions at the start of each exercise (e.g. describing the exercise, providing a model) and Pearson correlation coefficient for the number of behaviours elicited in each exercise. As the aim of the study was to determine the feasibility of the study design, we did not examine change using our clinical outcome measures37.

To assess acceptability of delivery of the therapy via Skype and acceptability of the study design we analysed transcribed interview data using an inductive thematic analysis38. One researcher (JS) repeatedly read and compared interview transcripts to develop codes to describe meaning within interviews, and grouped codes into over-arching themes and sub-themes. Verification of the codes and themes took place through discussion of code examples and potential themes with two other researchers (KB and LP). Disagreements were resolved through further review of the dataset and explanation of thinking behind particular coding or themes. Interview transcripts were coded and stored using Nvivo 10.

In the development of a tool to assess therapy costs we used descriptive statistics (median, range) to summarise data relating to therapists’ time spent on activities relating to each participant allocated to treatment as usual and completion of each section of the data collection form.

To determine if it is possible to reduce the amount of data collected, we examined the agreement between intelligibility measures taken on day one and day two of the paired recordings and between pre-therapy recordings using mixed effects regression models, with recording pair nested within data collection point, nested within child to obtain intra-class correlation coefficients39. We also used regression models to examine the effect of word list length on estimates of percentage intelligibility. Lists comprising the first 15, 20, 25 and 30 words were compared with the full 50 words. In the examination of the effect of word list length, we fitted child as a random effect and list length as a fixed effect. We used intraclass correlation coefficients to measure agreement between the reduced lists and the full 50 word list for the full dataset, and for pre-therapy and post-therapy data separately.

RESULTS

Feasibility

Recruitment

Thirty five children were identified and contacted about the study, of whom 22 were recruited (8 female, 14 male; mean age 8.8 years (SD 3.2)) between October 2014 and October 2015 (Figure 1). As dysarthria in cerebral palsy is a chronic condition and children remain on SLT caseloads throughout childhood, we examined identification of participants by services who had recruited children to previous studies separately from those with whom we had not worked before. Therapists from three Trusts who had identified participants in our previous research identified 1, 3 and 4 eligible children each, of whom 1, 2, 2 respectively were recruited. Therapists from five Trusts that had not collaborated in previous studies identified 3-5 (median =4) potential participants each, of whom 2-4 participated (median=3).

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All families who fitted the study criteria agreed to be randomised to Skype therapy or treatment as usual with the offer of Skype therapy at the end of the study, surpassing our feasibility criterion of 75%. Identification of the first participant in each of the first wave Trusts took 4-64 days and their recruitment took 19-45 days from identification.



Table 1 shows the characteristics of the participants in the two groups. There were more boys in the treatment as usual group, and overall this group had slightly lower receptive language and nonverbal understanding than the children allocated to the Skype therapy.


Skype dysarthria therapy group (n=11)

Usual therapy group (n=11)

Sex 5 M; 6 F 9 M; 2F



Dyskinetic 5

Spastic 6

Dyskinetic 3

Ataxic 2GMFCS median (IQR) III (I, IV) I (II, IV)

MACS median (IQR) II (I, III) II (II, III)

CFCS median (IQR) III (I, IV) II (II, III)

Viking Speech Scale median (IQR) III (II, III) III (II, III)

Mean language comprehension age (TROG2 age equivalent score)

7.8 (3.3) 5.6 (2.9)

Mean nonverbal understanding age (Raven’s Progressive Matrices age equivalent score)

6.6 (2.8) 4.9 (2.0)



GMFCS: Gross Motor Function Classification Scale40; MACS: Manual Ability Classification Scale41; CFCS Communication Function Classification System42; TROG2; Test of Reception of Grammar 2nd edition31; DEAP: Diagnostic Examination of Articulation and Phonology

Attrition

None of the participants withdrew from the study. All children allocated to Skype dysarthria completed six weeks of intervention. All children remained in the arm to which they were originally

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allocated, surpassing our feasibility criterion of 75%. However, due to the deadline set by our funding body, four families were unable to complete measures at twelve weeks post therapy.


We completed 206 of 220 planned recordings (94%) of children’s speech. Seven recordings, from the last four children recruited, could not be completed by the cut-off for recordings which was imposed by the research time frame. A further 7 recordings (from four children) could not take place due to child illness (4) or holidays (3). We recruited 213 listeners over 12 weeks, who rated a mean of three recordings each (SD 0.15). Parent and teacher compliance with questionnaire (FOCUS) completion was low: 29/44 parent questionnaires (66%); 26/44 teacher (59%); 34 pre-therapy (77%); 22 post-therapy (50%).

Table 2 provides a breakdown of measures collected for participants in the Skype dysarthria and usual therapy groups.

Table 2 Outcome measures completed for participants in the Skype dysarthria usual therapy groups

Measure Skype dysarthria therapy group

Usual therapy group


6 week pre therapy

1 week pre therapy

1 week post therapy



22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

17/18 (94.4)

22/22 (100)

21/22 (95.5)

20/22 (90.9)

22/22 (100)

19/19 (94.4)Number of listeners hearing recordings / total possible (%)

6 week pre therapy

1 week pre therapy

1 week post therapy



57/66 (86.4)

62/66 (93.9)

59/66 (89.4)

58/66 (87.9)

48/54 (88.9)

58/66 (87.9)

61/66 (92.4)

60/66 (90.9)

60/66 (90.9)

53/57 (93.0)Number of FOCUS questionnaires completed / total possible (%)

Parents pre therapy

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8/11 (72.7)

6/11 (54.5)

8/11 (72.7)

6/11 (54.5)

10/11 (90.9)

5/11 (45.5)

8/11 (72.7)

4/11 (36.4)Number of parents rating therapy effectiveness / total possible (%)

1 week post therapy



11/11 (100)

10/10 (100)

9/9 (100)

NA

NA

NA


Children received a mean of 15 sessions (SD 2.7, median 16, range 11-18). Children missed sessions because of illness and competing family commitments. Internet connection issues were encountered in 32/147 sessions (median 2, range 0-10 per child). Therapy time lost to connection problems ranged from 2-30 minutes per child over their programme of therapy, (median 15; range 0-38). Longer connection issues were encountered when new devices were used and one session was abandoned due to Skype issues that affected users globally.

Video recordings of 31 Skype dysarthria therapy sessions (19%) were checked for treatment fidelity. Agreement between the two raters on the presence of therapy activities at the start of each exercise was substantial43 (Κ=0.72) and high for the numbers of behaviours produced in each exercise (r =0.99, p<0.001). Twenty sessions (65%) met the criterion of 120 behaviours (mean 121, range 74-140), thus failing to meet the feasibility criterion of 85% of sessions containing 120 speech behaviours. Failure to elicit 120 speech behaviours arose because the functional phrases selected by five participants for practice in Exercise 2 were long and could not be spoken on one breath. This curtailed the time left for rapid elicitation of novel words and phrases and random practice in Exercises 3 and 4. In two sessions Skype connection difficulties reduced time available to complete the exercises.


All eight children and eight parents who rated effectiveness of therapy at twelve weeks post intervention deemed it at least moderately effective (median rating by children 5, range 5-7; median rating by parents 6, range 4-7). Should the three children and their parents who were unable to complete ratings due to time limitations have judged it less than somewhat effective (i.e. 8/11 (72.7%) judging therapy as effective), the criterion of 67% rating the therapy as at least somewhat effective would have been reached.

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Acceptability

Skype delivery of therapy

Discussion about the delivery of the intervention via Skype centred around four key themes: 1) Familiarity with technology; 2) Remote connections; 3) Fit with family life and 4) Unforeseen benefits.

Familiarity with technology

During the interviews none of the parents raised concerns about using Skype for therapy. Those allocated to the Skype therapy described themselves as being familiar with using technology for communication; some had used Skype before to keep in touch with relatives and others had used alternative video communication software such as Facetime. Parents were happy for the children to be contacted by the research therapists via Skype for pre-arranged sessions once they had accepted the study Skype name as a contact.

Skype I haven’t used in probably about five or six years. But yes, we’ve used FaceTime a lot. (Father A)

I'll show you how to get Skype on and then you'll get that ready. When I tell him, once he's done it, he'll know how to set it up because he's good with computers, so it won't be a problem. (Mother E)

Children were also positive about therapy via their tablet/computer, seeing it as ‘cool’ (Child G). Most appeared confident about using the technology from the start, although one child who had not used Skype or FaceTime had lots of questions about how to work the software and appeared a little anxious about being able to connect. However, in post therapy interviews this child reported that she quickly ‘got used’ to Skype after the first couple of sessions.

I know Skype. Will it ring? (Child Q)

There is a button you have to press, you know the ‘enter’ button on the computer? When you’ve got the little arrow, and you can move the arrow around with the mouse? (Interviewer)

I don’t know, yes I do. And you just click the button. What if we don’t have a mouse? I think it’s going to be better if someone like peeked through the door. If I need help with a hard question and I peek through the door and say I am struggling, and they come and help me. (Child Q)

Remote connections

Parents reported that internet connections were generally good and that when connections were lost they were usually retrievable within a session.

There was one day I think [research therapist] was struggling to connect. She just Skyped a typed message saying, “I’m just having a few problems.” … But on the whole our Broadband

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has been fine because it does stick a little bit sometimes where we are. But we don’t seem to have had any issues really. (Mother E)

We never even had any issues with Wi-Fi connection and our Wi-Fi's rubbish. We never got a lost signal through once, I don't think that's happened once. If there is any, it's just like me being the idiot and not turning up the volume up. (Father A)

Parents and children reported enjoying therapy and feeling comfortable with the therapist.

Basically the same. Just on an iPad instead of face to face. (Child R)

I think it's great in the fact that it is over Skype. You've not got people coming in all the time, in and out of the house, which is not a problem. But it makes it a bit more fun for the youngsters, I think, by having Skype. [Child] was sitting there with his iPad and he was really enjoying it. (Mother R)

However, one child reported that sitting in the same position for the duration of the Skype sessions was difficult

It’s difficult to do sitting. On Skype it’s different. (Child V)

On Skype it’s difficult to sit still for that long. (Mother V)

Overall, although shorter sessions might be better for some children, it seemed that the families did not view the technology as a barrier to therapy.

Fit with family life

Parents and children reported that a key benefit of Skype therapy delivery was being able to have therapy at home; families did not have to rush to attend appointments or make additional car journeys, which are difficult for families of children with disabilities, making clinic appointments stressful for children, parents and siblings. Children liked doing therapy in familiar surroundings.

It’s nice that he can do it in his own home, just relaxing….it’s not like ‘ Right come on, we have to go out’ and drive there, in and out of his wheelchair…..it’s just another trip out which we would rather avoid if you don’t have to do it (Mother I)

However, the three times per week schedule was difficult to manage across the full six weeks for some families.

Yes it is manageable…. I couldn't say we could do a fourth one. Three is the limit. (Mother V)

Some parents thought that having some of the therapy sessions via the internet at school may facilitate greater engagement of education staff in children’s therapy and understanding of it.

I think that would be quite good because then, at least, you'd be able to get a bit more feedback from the school as to how they think that it's improved on. (Mother R)

Unforeseen benefits

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Parents reported that therapy delivery via Skype had an unforeseen benefit of promoting children’s independence. They described how children used their own computer or tablet, chose where to do therapy and some older participants logged on independently once parents were sure they recognised the Skype contact used by the researchers for the study. Parents described how they could remain nearby, see what was happening in therapy and be on hand to help if was required, but could also attend to other household tasks and spend time with their other children. This meant that parents could be involved but also let their children take control and ownership of their therapy.

I think she feels, it gives her a bit more independence. It's hers. …….We'll pop our head in now and again and make sure she's okay. She doesn't need us at all. She can just do it herself which is great. (Father A)

Yes, kind of, you’re listening to how well she’s doing, but equally taking in what the girls are doing with her, so that you can kind of apply some of it at home. (Mother G)

Research design

Discussion around the study design centred on the random allocation to Skype therapy or usual therapy and the outcome measures used in study. One of the motivating factors in taking part, even if allocated to usual care, was the offer of therapy via skype at the end, and this seemed crucial for successful engagement with participants. :

I think if it was just for a study [without offer of Skype therapy], I think we would have been less likely to (participate). (Father D)

That was the great thing, it didn’t matter when, you were going to get it regardless, so it was worth it. (Mother O)

The visit schedule for the collection of data was time consuming, but deemed to be manageable for the duration of the study.

It is a big commitment, yes. If you've got the time, they do make you aware of what time you're going to need prior to the therapy. So it's not like you're not aware of it. (Mother R)

Just the fact that everything has to be quiet, that’s quite tricky with having a three year old who’s quite noisy; that was quite hard. And the fact that it had to be all in one week as well. That was quite tricky. But certainly I wouldn’t say that it was a negative thing, it just was more difficult to fit in really. (Mother V)

All families allocated to treatment as usual accepted the offer of Skype dysarthria therapy at the end of the study. Having therapy immediately after the end of the study period was seen as preferable for both parents and children allocated to treatment as usual

It’s going to work out that we’ve had continuous visits, regular visits, and then the therapy will start for them. It’s actually maybe worked out quite well. It’s not like it’s been forgotten and then, “Oh,” all of a sudden here’s the therapy from something you did six months ago. (Mother F and J)

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All parents and all participants reported that having multiple speech recordings made for the study was acceptable, but that the task did become boring. One child reported that she didn’t like being recorded but quickly got used to it.

I’m used to it now. It’s not hard for me, it’s easy now. It used to be hard when I first started. Now it’s getting easier and easier. (Child C)

Neither participants nor parents reported any concerns about the recordings being heard by strangers in order to estimate children’s intelligibility.

You’ve got to measure the improvement…. There’d be no point in doing it if you weren’t going to see a difference….. So it has to be somebody who doesn’t know the children or who have never heard them before” (Mother E)

Most parents also reported that the FOCUS questionnaire was relevant and easy to complete, and none reported reasons for not returning the data. One mother reported that she had to check the meaning of some items and was concerned that she couldn’t recall previous ratings and wanted to report that she thought her child had improved.

(remember thinking) I’m misinterpreting this. I can’t just fathom out what they’re asking me. …. I should have taken a photocopy actually of my answers. Just to remember what I’d put last time (Mother E)

Harms


Economic measures of therapy costs

Therapists of all children allocated to treatment as usual and the research therapists completed all sections of the resources questionnaire. Results suggested that some sections could be simplified (therapists’ travelling time to see children could be left open for therapists to complete).

Six children allocated to the treatment as usual group received at least one session (median 1; range 0-7) of speech and language therapy from NHS services in the six weeks therapy period assessed for the study. Of the six children who did receive input, two were seen for single review sessions only, two received regular therapy focussed on expressive language and two received therapy targeting articulation. Therapy sessions were usually 31-40 minutes in duration. Staff salary bands ranged from 4 (therapy assistant) to 7 (specialist).

The literature review identified 13 articles reporting the use of tools previously used in this client group to estimate health state utilities. Apart from generic tools developed for use in children e.g. HUI2©, EQ-5D-Y, no other suitable tools were identified. A further tool the CHU-9D, whilst not previously used, was also considered potentially suitable.


There were strong intra-class correlations between percentage intelligibility across pairs of recordings within the same subject at each of the five time points, for single words this was 0.87 (95% CI 0.79, 0.91) and connected speech 0.82 (95% CI 0.73, 0.89). Intraclass correlations were also

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strong between recordings taken at one and six weeks before therapy for single words (0.83, 95% CI 0.7, 0.90) and connected speech (0.73, 95% CI 0.57, 0.86).

Mixed effect regression models showed very little impact of list length on intelligibility after adjusting for the random effect of child when the full 50 item lists were compared with 30, 25 or 20 items. However, when the first 15 items were compared with the full 50 item list a significant difference was observed (95% CI -3.87, -0.36). There were strong intra-class correlations between the 20, 25, 30 and 50 words of the CSIM lists , for both the full dataset and when pre-therapy and post therapy data were examined separately (Table 3).

Table 3 Mean and standard deviation of percentage of words understood in the first 15, 20, 30 and 40 words and full 50-word lists from the Children’s Speech Intelligibility Measure

Comparison with full 50 word listPercentage of words perceived correctly by

listenersAll data Pre-therapy Post-therapy

Words in CSIM lists

Mean (sd)

95% CI ICC 95% CI ICC 95% CI ICC 95% CI

Full 50 word list

50.27 (18.51)

48.82-51.72 - - - - - -

First 30 item

49.78 (19.41)

48.26-51.30 0.97 0.96-0.97 0.97 0.95-0.98 0.97 0.96-0.98

First 25 items

49.67 (19.84)

48.11-51.22 0.96 0.95-0.96 0.96 0.94-0.97 0.96 0.95-0.97

First 20 items

49.55 (20.25)

47.96-51.13 0.93 0.93-0.94 0.92 0.90-0.94 0.94 0.92-0.95

First 15 items

48.45 (21.14)

46.80-50.11 0.89 0.87-91 0.87 0.83-0.90 0.90 0.87-0.92

DISCUSSION

Summary of findings

This study aimed to test the feasibility of conducting a randomised controlled trial of the clinical and cost effectiveness of intensive dysarthria therapy versus usual treatment for children with cerebral palsy. Results suggest that a randomised controlled trial is feasible and acceptable but that the study processes and data collection can be simplified to reduce research costs and burden to participants.

The study design was acceptable to families. Families joined the research because they were seeking therapy to improve children’s speech intelligibility and communicative independence, which is highly prized44. An important component of feasibility of recruitment was the offer of the experimental therapy to families allocated to treatment as usual at the end of data collection, even though the treatment may turn out not be effective. Our findings here concur with the results of previous trials of interventions for children with other types of neurodisability, such as autism45-47, and support the conclusion that parents of children with neurodisability often feel the need to ‘do more’ for their

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children continue to seek additional input that could aid their child’s development48, 49. There was no loss of precision in estimates of variation of intelligibility when we reduced the number of words assessed from 50 to 20, which is similar to other tests of intelligibility50. Future trials could limit data collected to single recordings of 20 single words and picture description at one week pre therapy, and one, six and twelve weeks post therapy. Return of the FOCUS questionnaire was low (approximately 60%) and could possibly be improved if usual mechanisms of communication (e.g. home-school diaries and bags) were used51.

Similar to other studies of teletherapy for children with speech disorders and adults with dysarthria22, 24, 52, interviews with parents and participants suggest that provision of therapy via Skype is feasible and acceptable to families. Therapy was delivered according to the therapy protocol and children and parents reported no difficulties in maintaining rapport with the therapist, supporting other recent research22, 24, 53 One possible advantage of the study design was that the research therapists were familiar to families by the time therapy was due to commence, having carried out the assessments and speech recordings. Shaw and colleagues, in their study of general practice consultations, found that sessions via video-conferencing flowed more easily when patients and practitioners knew each other and had time to build a relationship53. Internet connection issues affected one in five sessions in our study, but were generally short and resolvable as previous UK research53. However, connection difficulties affected some children more than others, possibly due to broadband speed. Test sessions to establish Skype connection with families and to confirm computer settings would be advisable in future research and if implementing Skype in therapy53. None of the parents reported any concerns about internet safety and were happy for therapists to contact the family via Skype to start therapy sessions. Individualised actions to maximise internet safety would be required in a trial, as recommended for all children54, and Trusts would need to assess information governance issues individually53. However, Skype is now being piloted by NHS Trusts as a medium to conduct consultations and advice exists on its use in clinical settings.55-57

For most children it was possible to complete all therapy exercises within 40 minutes. Problems that occurred were due to a selecting phrases that were too long to say in one breath. A similar finding has been noted for adults with dysarthria caused by Parkinson’s disease58. This could be readily avoided in future trials. Shortening the phrases may also help to reduce the overall session length, making them more accessible to children who find it difficult to maintain their concentration.

Therapy was conducted by three generalist clinicians who had no experience of childhood motor speech disorders or teletherapy. As such, they were similar to many NHS therapists who provide services to children in mainstream schools in the UK, but less experienced than therapists who provide specialist services to children with motor disorders15. This study has shown that the protocol can be followed by junior therapists and by extension it should be suitable for therapists with greater experience. Future trials should test the effectiveness of the therapy when delivered via local therapists, and include a process evaluation to evaluate barriers to and facilitators of implementation which have not been identified in our research to date59. If a study were clustered at the unit of Trust, to avoid contamination between therapists providing intervention and participants interacting with each other, mean cluster size can be estimated as three, based on recruitment from Trusts who had not participated in previous studies. We recruited a small number of participants who were known to NHS speech and language therapy services via parent networks and support organisations. Advertisement of trials via parental support networks should be encouraged to reduce gatekeeping.

Limitations

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This study recruited participants from eight trusts within one region. Recruitment may not be representative of Trusts in other parts of the UK. However, to our knowledge no research is being conducted on children’s speech elsewhere in the UK and there is sparse research internationally20. Families participating in this study used their own laptops/tablets for the remote therapy and may represent a ‘tech-savvy’ group. Teletherapy in NHS is only feasible to families with a computer and internet access; consideration would be needed as to whether this approach might widen socio-economic health inequalities.

Intelligibility measures were conducted by the research therapists, which could have prompted children to use their new voice during measurement. Future research should ensure that measurements are conducted by personnel who have not been involved in therapy but who have built up a rapport with children so that they feel comfortable during in the assessments.

All parents rated speech to have improved at least moderately by twelve weeks. Such lack of variation precludes the calculation of a minimally important difference in speech intelligibility using anchor-based methods and suggests change in percentage intelligibility should be used to inform sample size calculation.60 Only one child judged their speech to have improved moderately (level 4); all others judged their speech to be a (very) great deal better (6 or 7) at all time points. High ratings may be due to response bias and social desirability. However, children may have experienced change that led them to report their speech being a (very) great deal better. Furthermore, they may also have developed confidence in or from their ability to be understood, as reported in the interviews. Future research must measure the impact of therapy on children’s participation and well-being as well as their intelligibility.13

The aim of usual treatment and a very brief description of the intervention was provided by local therapists in our costing tool. Although the tool showed that none of the children in the treatment as usual group received intervention focussing on breath support, phonation and rate to increase the clarity of children’s speech, it did not enable us to describe the intervention that the children received. For example, we do not know if motor learning principles that underpinned the Skype dysarthria therapy were included in articulation therapy. Future research should include not only the aim and focus of treatment as usual but also the active ingredients. The TIDieR checklist61 could be used as a basis for expanding the tool.

CONCLUSIONS

The study suggests that a randomised controlled trial of dysarthria therapy is feasible, that therapy can be provided via Skype to children with cerebral palsy and that families would participate in a trial if all arms of the study included provision of Skype dysarthria therapy at some point. Future trials could use a smaller data set to measure intelligibility than previously tested, which would reduce the time and costs required to conduct a trial and reduce the burden of participating in the study for families.

FUNDING STATEMENT

This paper presents independent research funded by the National Institute for Health Research (NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-0712-28077). The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health and Social Care.

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ETHICS STATEMENT The study was approved by National Research Ethics Service Committee Leeds East Research Ethics Committee (14/YH/0058).


LP, ES, LV, KB designed the study. LP coordinated the study and took overall responsibility for the research. MO developed the website for conducting listening tasks. HK, NP, JS, KS and PA had responsibility for data collection. HK, NP, KS delivered the therapy. LP, ES, PA, LV, KB, JS analysed data. LP drafted the paper. All authors have contributed to and approved the final version of the paper.





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REFERENCES

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Research Support, Non-U.S. Gov't. DOI: http://dx.doi.org/10.1111/j.1469-8749.2009.03366.x.19. Boliek C and Fox C. Therapeutic effects of intensive voice treatment (LSVT LOUD®) for children with spastic cerebral palsy and dysarthria: A phase I treatment validation study. International Journal of Speech-Language Pathology 2017; 19: 601-615. DOI: 10.1080/17549507.2016.1221451.20. Pennington L, Parker NK, Kelly H, et al. Speech therapy for children with dysarthria acquired before three years of age. Cochrane Database of Systematic Reviews 2016: Art. No.: CD006937. DOI: 006910.001002/14651858.CD14006937.pub14651853.21. Fox C and Boliek C. Intensive voice treatment (LSVT LOUD) for children with spastic cerebral palsy and dysarthria. Journal of Speech, Language, and Hearing Research 2012; 55: 930-945.22. Wales D, Skinner L and Hayman M. The Efficacy of Telehealth-Delivered Speech and Language Intervention for Primary School-Age Children: A Systematic Review. International Journal of Telerehabilitation 2017; 9: 55-70. DOI: 10.5195/ijt.2017.6219.23. Grogan-Johnson S, Schmidt AM, Schenker J, et al. A Comparison of Speech Sound Intervention Delivered by Telepractice and Side-by-Side Service Delivery Models. Communication Disorders Quarterly 2013; 34: 210-220.24. Fairweather GC, Lincoln MA and Ramsden R. Speech-language pathology teletherapy in rural and remote educational settings: Decreasing service inequities. International Journal of Speech-Language Pathology 2016; 18: 592-602. DOI: 10.3109/17549507.2016.1143973.25. Molini-Avejonas DR, Rondon-Melo S, Amato CAdLH, et al. A systematic review of the use of telehealth in speech, language and hearing sciences. Journal of Telemedicine and Telecare 2015; 21: 367-376. DOI: 10.1177/1357633x15583215.26. Pennington L, Smallman CE and Farrier F. Intensive dysarthria therapy for older children with cerebral palsy: findings from six cases. Child Language Teaching and Therapy 2006; 22: 255-273. DOI: http://dx.doi.org/10.1191/0265659006ct307xx.27. Julious SA. Sample size of 12 per group rule of thumb for a pilot study. Pharmaceutical Statistics4 2005; 4: 287 – 291.28. Pennington L, Virella D, Mjøen T, et al. Development of The Viking Speech Scale to classify the speech of children with cerebral palsy. Research in Developmental Disabilities 2013; 34: 3202-3210.29. Dodd B, Hua Z, Crosbie S, et al. Diagnostic Evaluation of Articulation and Phonology Oxford: Pearson Assessment, 2006.30. Hayden D and Square P. Verbal motor production assessment for children. San Antonio: Harcourt Assessment, 1999.31. Bishop DV. Test of Reception of Grammar 2. Oxford: Pearson Assessment, 2003.32. Raven JC. Raven’s Coloured Progressive Matrices. San Antonio, Tx: PsychCorp, 1998.33. Wilcox K and Morris S. Children's speech intelligibility measure. San Antonio: Harcourt Assessment, 1999.34. Thomas-Stonell N, Oddson B, Robertson B, et al. The FOCUS: Focus on the Outcomes of Communication Under Six, http://www.hollandbloorview.ca/research/FOCUS/documents/Parent_FOCUS_B_rev_13.pdf (2012).35. Guyatt GH, Osoba D, Wu AW, et al. Methods to explain the clinical significance of health status measures. Mayo Clinic Proceedings 2002; 77: 371-383.36. Rauch R, Smith J, Brittain K, et al. Intensive speech therapy for children with cerebral palsy: the views of children and parents on effectiveness and acceptability. submitted.37. Arain M, Campbell MJ, Cooper CL, et al. What is a pilot or feasibility study? A review of current practice and editorial policy. BMC Medical Research Methodology 2010; 10.38. Braun V and Clark V. Using thematic analysis in psychology. Qualitative Research in Psychology 2006; 3: 77-101.39. Streiner DL and Norman GR. Health measurement scales: a practical guide to their development and use. 3rd edition. 3 ed. Oxford: Oxford University Press, 2003.

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40. Palisano R, Rosenbaum P, Bartlett D, et al. Gross Motor Function Classification System Expanded and Revised. Toronto: CanChild Centre for Childhood Disability Research, McMaster University, 2007.41. Eliasson A, Krumlinde Sundholm L, Rösblad B, et al. The Manual Ability Classification System (MACS) for children with cerebral palsy: scale development and evidence of validity and reliability. Developmental Medicine & Child Neurology 2006; 48: 549-554.42. Hidecker MJC, Paneth N, Rosenbaum PL, et al. Developing and validating the Communication Function Classification System for individuals with cerebral palsy. Developmental Medicine and Child Neurology 2011; 53: 704-710.43. Landis J and Koch G. The measurement of observer agreement for categorical data. Biometrics 1977; 33: 159-174.44. Morris C, Janssens A, Allard A, et al. Informing the NHS Outcomes Framework: evaluating meaningful health outcomes for children with neurodisability using multiple methods including systematic review, qualitative research, Delphi survey and consensus meeting. Health Service Delivery Research 2014; 2.45. McConachie H, Randle V, Hammal D, et al. A controlled trial of a training course for parents of children with suspected autism spectrum disorder. Journal of Pediatrics 2005; 147: 335-340.46. McConachie H, McLaughlin E, Grahame V, et al. Group therapy for anxiety in children with autism spectrum disorder. Autism 2014; 18: 723-732. DOI: 10.1177/1362361313488839.47. Grahame V, Brett D, Dixon L, et al. Managing repetitive behaviours in young children with Autism Spectrum isorder (ASD): Pilot randomised controlled trial of a new parent group intervention. Journal of Autism and Developmental Disorders 2015; 45: 3168-3182. journal article. DOI: 10.1007/s10803-015-2474-x.48. Beresford B, Clarke S and Maddison J. Therapy interventions for children with neurodisabilities: a qualitative scoping study. Health Technology Assessment 2018; 22.49. Marshall J and Goldbart J. 'Communication is everything I think.' Parenting a child who needs Augmentative and Alternative Communication (AAC). International Journal of Language and Communication Disorders 2008; 43: 77-98.50. Hodge M, Daniels J and Gotzke CL. TOCS+ Intelligibility Measures [computer software]. Edmonton, Canada: University of Alberta, 2009.51. Dobbins M and Abbott L. Developing partnership with parents in special schools: parental perspectives from Northern Ireland. Journal of Research in Special Educational Needs 2010; 10: 23-30. DOI: doi:10.1111/j.1471-3802.2009.01140.x.52. Constantinescu G, Theodoros D, Russell T, et al. Treating disordered speech and voice in Parkinson's disease online: A randomized controlled non-inferiority trial. International Journal of Language & Communication Disorders 2011; 46: 1-16.53. Shaw S, Wherton J, Vijayaraghavan S, et al. Advantages and limitations of virtual online consultations in a NHS acute trust: the VOCAL mixed-methods study. Health Service Delivery Research 2018; 6.54. (UKCCIS) UCfCIS. Advice on child internet safety 1.0 Universal guidelines for providers https://www.gov.uk/government/uploads/system/uploads/attachment_data/file/251455/advice_on_child_internet_safety.pdf (2012, accessed 13 September 2017).55. Bostock N. GP pilots Skype consultations with patients. GP Online. 2013.56. NHS England. https://www.england.nhs.uk/2016/01/newham-diabetes-pilot-scheme/ (2016, accessed 13 September 2017).57. QIPP Digital Technology. A brief guide to Skype remote consultations, https://www.networks.nhs.uk/nhs-networks/qipp-digital-technology-and-vision/documents/QIPP%20DT%20Skype%20Factsheet%20v1.pdf (2012, accessed 13 September 2017).58. Theodoros DG, Hill AJ and Russell TG. Clinical and Quality of Life Outcomes of Speech Treatment for Parkinson's Disease Delivered to the Home Via Telerehabilitation: A Noninferiority

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Randomized Controlled Trial. American Journal of Speech-Language Pathology 2016; 25: 214-232. DOI: 10.1044/2015_AJSLP-15-0005.59. Craig P, Dieppe P, MacIntyre S, et al. Developing and evaluating complex interventions: New guidance. London: Medical Research Council, 2008.60. Cook JA, Hislop J, Altman DG, et al. Specifying the target difference in the primary outcome for a randomised controlled trial: guidance for researchers. Trials 2015; 16: 1-7. journal article. DOI: 10.1186/s13063-014-0526-8.61. Tammy CH, Paul PG, Isabelle B, et al. Better reporting of interventions: template for intervention description and replication (TIDieR) checklist and guide. BMJ 2014; 348. DOI: 10.1136/bmj.g1687.

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Figure 1. CONSORT flow diagram

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297x420mm (300 x 300 DPI)

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Internet delivery of intensive speech and language therapy for children with cerebral palsy

Rating of effectiveness of therapy Parents ‘Is your child’s speech better, about the same or worse than before they started the Skype speech therapy?’ (if better) ‘How much better is your child’s speech? ’

1 2 3 4 5 6 7 Almost the same A little better Somewhat better Moderately better A good deal better A great deal better A very great deal

better If worse ‘How much worse is your child’s speech?’

1 2 3 4 5 6 7 Almost the same A little worse Somewhat worse Moderately worse A good deal worse A great deal worse A very great deal worse Children ‘Is your speech better, about the same or worse than before they started the Skype speech therapy?’ (if better) ‘How much better is your speech? ’

1 2 3 4 5 6 7

If worse ‘How much worse is your speech?’

1 2 3 4 5 6 7

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1


FIDELITY TREATMENT INTEGRITY CHECK©

INTENSIVE SPEECH AND LANGUAGE THERAPY FOR CHILDREN WITH CEREBRAL PALSY:

A SYSTEMS APPROACH THERAPY PROTOCOL (ROBSON, MILLER, PENNINGTON 207)

Participant SLT

Date of session

Observer Date of observation

1) TARGET VOICE IN SUSTAINED VOWEL

Production of /a:/



3. Therapist describes exercise: practice ‘ah’ 10 times YES/NO


5. Therapist instructs child e.g. “Copy me/you do it” YES/NO

6. Therapist provides stimulus phrase e.g. “use your [target voice name] voice YES/NO

7. Number of repetitions elicited (/10)

2) OPTIMUM SPEECH LOUDNESS AND RATE

Production of functional phrases





12. Therapist instructs child e.g. “You do it/Your turn” YES/NO

13. Therapist provides stimulus phrase e.g. “use your [target] voice” YES/NO

14. 3 repetitions of each functional phrase elicited YES/NO






3) STIMULATE OPTIMAL LOUDNESS IN NOVEL STIMULI

The child is asked to repeat 80 - 120 stimuli provided by the therapist. Circle stage

Stage a) Single words or phrases Stage b) Phrases Stage c) Extended speech Stage d) Increased cognitive difficulty - describe Stage e) Conversation


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2




20. Therapist provides elicitor appropriate to phase YES/NO

21. Therapist provides stimulus phrase e.g. “use your ‘nice and easy’ voice” YES/NO

22. Number of behaviours elicited (/80)






4) RANDOMISATION OF STIMULI



27. Therapist describes exercise: practice of all targets YES/NO


29. Number of items elicited (/20)

30. Feedback





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CONSORT 2010 checklist of information to include when reporting a pilot or feasibility trial*


Reported on page No

Title and abstract

1a Identification as a pilot or feasibility randomised trial in the title 1

1b Structured summary of pilot trial design, methods, results, and conclusions (for specific guidance see CONSORT abstract extension for pilot trials)

2

Introduction

Background and

objectives

2a Scientific background and explanation of rationale for future definitive trial, and reasons for randomised pilot trial

3

2b Specific objectives or research questions for pilot trial 3

Methods

Trial design 3a Description of pilot trial design (such as parallel, factorial) including allocation ratio 4

3b Important changes to methods after pilot trial commencement (such as eligibility criteria), with reasons NA


4b Settings and locations where the data were collected 4 - 7

4c How participants were identified and consented 5



5

Outcomes 6a Completely defined prespecified assessments or measurements to address each pilot trial objective specified in 2b, including how and when they were assessed

8

6b Any changes to pilot trial assessments or measurements after the pilot trial commenced, with reasons NA

6c If applicable, prespecified criteria used to judge whether, or how, to proceed with future definitive trial 8

Sample size 7a Rationale for numbers in the pilot trial 4


Randomisation:

Sequence

generation


8b Type of randomisation(s); details of any restriction (such as blocking and block size) 6

Allocation

concealment

mechanism

9 Mechanism used to implement the random allocation sequence (such as sequentially numbered containers), describing any steps taken to conceal the sequence until interventions were assigned

6

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interventions

6

Blinding 11a If done, who was blinded after assignment to interventions (for example, participants, care providers, those


6

11b If relevant, description of the similarity of interventions NA

Statistical methods 12 Methods used to address each pilot trial objective whether qualitative or quantitative 8

Results

Participant flow (a

diagram is strongly

recommended)

13a For each group, the numbers of participants who were approached and/or assessed for eligibility, randomly assigned, received intended treatment, and were assessed for each objective

9 and Figure

13b For each group, losses and exclusions after randomisation, together with reasons 10 and Figure


14b Why the pilot trial ended or was stopped NA

Baseline data 15 A table showing baseline demographic and clinical characteristics for each group Table 1

Numbers analysed 16 For each objective, number of participants (denominator) included in each analysis. If relevant, these numbers

should be by randomised group 11, 12

Outcomes and

estimation

17 For each objective, results including expressions of uncertainty (such as 95% confidence interval) for any estimates. If relevant, these results should be by randomised group

12, 12, 16

Ancillary analyses 18 Results of any other analyses performed that could be used to inform the future definitive trial 12-15


19a If relevant, other important unintended consequences 14

Discussion

Limitations 20 Pilot trial limitations, addressing sources of potential bias and remaining uncertainty about feasibility 18

Generalisability 21 Generalisability (applicability) of pilot trial methods and findings to future definitive trial and other studies 18

Interpretation 22 Interpretation consistent with pilot trial objectives and findings, balancing potential benefits and harms, and

considering other relevant evidence 17, 19

22a Implications for progression from pilot to future definitive trial, including any proposed amendments 17

Other information

Registration 23 Registration number for pilot trial and name of trial registry NA

Protocol 24 Where the pilot trial protocol can be accessed, if available Not available


26 Ethical approval or approval by research review committee, confirmed with reference number 19

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Citation: Eldridge SM, Chan CL, Campbell MJ, Bond CM, Hopewell S, Thabane L, et al. CONSORT 2010 statement: extension to randomised pilot and feasibility trials. BMJ. 2016;355.

*We strongly recommend reading this statement in conjunction with the CONSORT 2010, extension to randomised pilot and feasibility trials, Explanation and Elaboration for important

clarifications on all the items. If relevant, we also recommend reading CONSORT extensions for cluster randomised trials, non-inferiority and equivalence trials, non-pharmacological

treatments, herbal interventions, and pragmatic trials. Additional extensions are forthcoming: for those and for up to date references relevant to this checklist, see www.consort-statement.org.

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